Schistosoma mansoni. The adult parasites inhabit the blood vessels of the hepatic portal system of the main host. The disease may range from being asymptomatic to provoking liver damage or portal hypertension. Furthermore, ectopic schistosomiasis may develop, and several hypotheses have been raised to explain the occurrence of the disease. This paper describes two cases, one in a 39-year-old woman and the other in a 47-year-old woman. Both had similar symptoms of pain and abdominal distension caused by a large abdominal/pelvic mass. Histopathology of the ovary showed a mucinous cystadenocarcinoma of the intestinal type in the first patient and a papillary serous carcinoma in the second, with both tumors containing viable eggs of Schistosoma mansoni. The neoplasms probably serve as a migratory route for the adult parasites and the embolization of eggs. Nevertheless, there is insufficient evidence to confirm the malignization of a benign lesion due to the presence of Schistosoma mansoni. Few cases have been reported in the international literature on the association between ovarian schistosomiasis and neoplasms.Schistosomiasis is a parasitic infection that is highly prevalent worldwide, with a variety of species being responsible for causing the disease. In Brazil, however, the only identified species is  Ovarian epithelial carcinoma is the second most common cancer of the female genital tract. It corresponds to 5% of all female tumors  and is tProtective factors for ovarian cancer include parity, breastfeeding, the use of oral contraceptives, late menarche, early menopause, and surgical procedures such as hysterectomy and salpingectomy. Risk factors include the use of ovulation-inducing drugs, high estrogen and androgen levels, and genetic and environmental factors .In rare cases, an association has been reported between neoplasms and schistosomiasis, with studies showing that the ovaries are affected in only 0.5% of cases of ectopic schistosomiasis . S. mansoni is endemic in the Caribbean islands, Middle East, South America, and Africa and can be imported to any region of the world through immigration [ S. mansoni worldwide. In Brazil, this figure is estimated at 4 to 6 million individuals [ Schistosoma cause schistosomiasis; however, only S. mansoni has been reported in Brazil [Schistosomiasis caused byigration . The disigration . Betweenividuals . Currentividuals . Variousn Brazil .This parasitic infection is an important cause of morbidity and mortality and has been found in various organs of the body. There is generally no clinical suspicion of schistosomiasis in these lesions; indeed, in the majority of cases, the infection is only discovered by chance at histopathology . Schistosoma mansoni (S. mansoni) eggs associated with an ovarian mucinous cystadenocarcinoma of the intestinal type (case #1) and with a papillary serous carcinoma (case #2) in patients receiving care at the Department of Obstetrics and Gynecology, Santa Casa de Misericórdia Hospital, Vitória, Espírito Santo, Brazil. The internal review board approved this report under reference number 25471214.0.0000.5065. Both patients gave their signed informed consent to the publication of the images that form part of this paper.Two cases are reported here in which histopathology identified viableThis patient, a 39-year-old woman, was hospitalized complaining of progressive abdominal distension and edema of the lower limbs over the previous month. She also complained of abdominal pain and frequent micturition. There were no signs of dysuria, gastrointestinal symptoms, fever, loss of appetite, or weight loss.Physical examination showed the patient to be in an apparently good general state of health. She was well hydrated, her temperature was normal, and there were no signs of anemia, jaundice, or cyanosis. No cardiovascular or respiratory abnormalities were found. The distended abdomen presented normal bowel sounds, with a large, hardened, irregular mass extending from the pelvis to the epigastrium. Palpation of the liver and spleen revealed no abnormalities. There were no signs of ascites or enlarged lymph nodes. Gynecological examination revealed an apparently normal cervical epithelium and external os with no abnormal discharge. Digital pelvic examination showed a fibroelastic, closed cervix that was mobile and painless. Bimanual palpation revealed a large pelvic mass protruding into the Douglas pouch. The borders of the uterine fundus were unclear due to the size of the tumor.Pelvic ultrasonography revealed a large abdominal/pelvic mass of 6,606.7 cm³, with a predominantly cystic appearance and a solid component. There was an anechoic cystic mass of 4 cm³ in volume on the right ovary. Uterine volume was 180 cm³ and endometrial thickness was 6.7 mm. There were no signs of ascites. Doppler flow showed a low resistance index of 0.44.The results of supplementary tests showed CA-125 levels of 24.87 U/mL, hemoglobin 10.5 g/dL, hematocrit 35.6%, mean corpuscular volume (MCV) 81.2 fl, mean corpuscular hemoglobin (MCH) 27.5 pq, mean corpuscular hemoglobin concentration (MCHC) 32.2%, and coefficient of variation of red cell distribution width (RDW-CV) 12.0%. White blood cell count was 8,500/cm³, with 3% eosinophils (255/mm³).Exploratory laparotomy performed through a midline infraumbilical incision showed a large, predominantly cystic tumor occupying the entire abdominal/pelvic cavity, with its epicenter in the left ovary. Extraovarian lesions and signs of ascites were then noted. A hysterectomy was performed, with bilateral adnexectomy, partial omentectomy, and pelvic/aortic lymphadenectomy. The patient progressed satisfactorily with no postoperative complications and was discharged from hospital. S. mansoni eggs surrounded by eosinophilic granulomas permeating the neoplastic tissue were found in 2 of the 30 slides examined  83.2 fl, mean corpuscular hemoglobin (MCH) 28.5 pq, mean corpuscular hemoglobin concentration (MCHC) 34.2%, and coefficient of variation of red cell distribution width (RDW-CV) 13.0%. White blood cell count was 7,790/cm³, with 2% eosinophils (156/mm³).Exploratory laparotomy was performed through a midline infraumbilical incision, revealing a large pelvic mass of mixed composition occupying almost the entire abdominal/pelvic cavity, extending up to the hypochondrium, adhered to the uterus, adnexa, and bowel. Hysterectomy was performed and included bilateral adnexectomy, partial omentectomy, and pelvic/aortic lymphadenectomy. The patient progressed satisfactorily with no complications following surgery and was released from hospital. S. mansoni eggs were found  [The biological cycle of schistosomiasis is complex and well defined, with water being the means through which the parasite infects the main host (vertebrate/man). In turn, contaminated human feces in contact with water spread the infection to the intermediate host (invertebrate—thea snail) . The adua snail) .S. mansoni infestations are referred to as ectopic when the eggs or the adult form of the parasite is found outside the portal system, for example, in the skin, central nervous system, thyroid, myocardium, esophagus, stomach, gall bladder, pancreas, suprarenal, or urogenital tract [ S. mansoni eggs through the network of arteries as a consequence of the presence of congenital cardiovascular defects or arteriovenous shunts and (b) egg-laying after the migration of the helminthes [al tract . These pal tract . The mecal tract . Other mlminthes .The transfer of eggs to pelvic region may generate reactions in the internal genital organs, leading to dyspareunia, vaginal discharge, bleeding, dysmenorrhea, lower abdominal pain, or abdominal/pelvic tumors, all of these being nonspecific findings often reported by gynecologists . The vas S. mansoni eggs in the ovarian tumors described here strengthens this hypothesis. The presence of S. mansoni eggs causes inflammation and oxidative stress, which may lead to a poorer prognosis in the case of ovarian tumors. Nevertheless, there is insufficient evidence to confirm the malignization of a benign lesion due to the presence of S. mansoni. Further studies should be conducted in endemic areas to confirm this association, since in the majority of cases ectopic schistosomiasis lesions are diagnosed from surgical specimens or after death.Neoplastic processes such as the tumors described here produce vascular endothelial growth factor (VEGF) and fibroblast growth factor (FGF), amongst other substances that permit the proliferation, survival, and differentiation of the endothelial cells, thus generating neovascularization, which is fundamental for the survival and progression of the neoplastic cells . The neo S. mansoni and ovarian carcinoma are sparse in the literature; therefore, further studies should be conducted in endemic areas to provide further information on this association and on treatment in order to improve the current situation in which diagnosis in most cases is based on surgical specimens or reached following death.Studies on the association between


PEComas are a group of very rare mesenchymal neoplasms, which express myogenic and melanocytic markers, such as HMB-45 and actin. Situs inversus totalis represents a complete left to right side transposition of the asymmetrical thoracic and abdominal organs and incorporates dextrocardia. The presence of uterus PEComa in the setting of situs inversus totalis is extremely rare. Here, we report a case of PEComa of uterus with coexistence of situs inversus totalis and review the literatures. To the best of our knowledge this is the fist report of a uterus PEComa patient with situs inversus totalis. PEComas are a group of very rare mesenchymal neoplasms, which expresses myogenic and melanocytic markers, such as HMB-45 and actin. The PEComa family of tumors has subsequently grown to include angiomylipoma (AML), clear cell "sugar" tumor of the lung (CCST), lymphangioleiomyomatosis (LAM), and a number of unusual visceral, intraabdominal, soft tissue and bone tumors. Situs inversus totalis (SIT) represents a complete left to right side transposition of the asymmetrical thoracic and abdominal organs and incorporates dextrocardia. It is estimated to occur in between 1:5,000-20,000 adults.A 44-year-old woman visited our hospital reporting menorrhagia and dysmenorrhea with history of nausea and vomiting for weeks.Ultrasound scan showed the size of the uterus was 8.0 cm × 9.9 cm × 9.8 cm, the thickness of endometrium was 0.6 cm. There was inhomogeneous high echo with star-like color flow can be seen in the posterior wall of the uterus, and the range was 7.2 cm × 5.5 cm. The left ovary was 2.1 cm × 1.3 cm and the right was 2.6 cm × 1.8 cm, with the bilateral boundaries of the ovaries were clear. The bilateral fallopian tubes showed no obvious abnormalities.Ultrasound scan of abdomen showed the liver located on the left side of abdominal cavity, and the spleen located on the right side. The size and shape of the bilateral kidneys were normal.CT diagnosis report: The thoracic cage was symmetric. The bilateral lung fields were clear. The size of the heart was normal, dextrocardia. The abdominal organs were mirror reverse, situs inversus viscerum.Chest and abdominal computed tomography (CT) showed situs inversus totalis Fig. . A totalMacroscopically, the size of the uterus was 10 cm × 10 cm × 8.5 cm and there was an irregular nodular in the posterior wall of the uterus. The resected specimen of the nodular showed a white and gray, 7.0 cm in maximum diameter, an ill-defined border, and no capsule formation Fig. .Fig. 2ThThe tumor was fixed in 10 % formalin and embedded in paraffin. Several 4-μm sections were cut from each paraffin block. Hematoxylin-eosin (HE) and immunohistochemical (IHC) stains were performed. IHC staining was performed using the streptavidin-peroxidase system  according to the manufacturer's instruction. Commercially available prediluted monoclonal antibodies against the following antigens were employed: Vimentin ((V9), 1:200, Maxim), CK ((AE1/AE3), 1:200, Maxim), Melan-A((A103), 1:200, Dako), HMB-45, Desmin((D33), 1:200, Dako), SMA((1A4), 1:200, Maxim), ER((SP1), 1:200, Maxim) PR((SP2), 1:200, Maxim), p53((DO-7), 1:200, Maxim), Syn((SP11), 1:200, Maxim), chromogranin-A((DAK-A3), 1:200, Dako), S100((4C4.9), 1:200, Maxim), CD38((F7101), 1:200, Dako), CD138((M115), 1:200, Dako), and Ki-67 ((MIB-1), 1:200, Maxim). The immune reactions were visualized with the use of DAB as the chromogen . All internal and external controls worked appropriately.By histology, the tumor consisted of round and polygonal cells with clear to eosinophilic granular cytoplasm. The tumor cells proliferated in a honeycomb-like appearance and often were arranged in a radial fashion around blood vessels. The tumor cell nuclei showed hyperchromasia, nuclear enlargement, and small nucleoli Fig.  and b. TBy immunohistochemistry, the tumor cells were positive for Vimentin Fig. , Melan-AA uterus PEComa was diagnosed on the basis of the above findings. The patient received only surgical resection. She is undergoing regular physical examination and remains free of disease at 2 months after operation.The concept of the perivascular epithelioid cell (PEC) was proposed by Bonetti etal. in 1992 . PEComasThe differential diagnosis of soft tissue and gynecologic PEComas is fairly broad and predominantly guided by the morphology (epithelioid vs. spindled) and location of the tumor. Because of their expression of HMB-45 and/or melan-A, PEComas are frequently confused with both conventional melanoma and clear cell sarcoma. Some of the morphologic features of PEComas, including the admixture of spindled and epithelioid forms, the occasionally prominent nucleoli, and the presence of multinucleated cells, are also seen in melanoma and clear cell sarcoma. In most cases, melanoma/clear cell sarcoma can be distinguished from PEComa by their strong expression of S-100 protein and smooth muscle actin nonimmunoreactivity .Complete resection is the preferred treatment for PEComa at present, but for the specific scope of operation is no definite conclusion. Most authors believe that hysterectomy and adnexectomy are the most direct and effective means of treatment. But for the malignant PEComa of uterus, pelvic lymph nodes dissection and postoperative adjuvant chemotherapy remains controversial. The plan of chemotherapy is still in study and discussion.Situs inversus totalis (SIT) is a condition with left-to-right reversal of the viscera combined with dextrocardia and individual organs present a symmetrical mirror image morphology. It is frequently complicated by concurrent abnormalities in the cardiovascular system and the hepatobiliary system. The prevalence of SIT is estimated to occur in between 1:8000 and 1:25000 , 8. No rIn summary, we reported a unique case of PEComa of uterus with coexistence of situs inversus totalis and review the literatures. The presence of uterus PEComa in the setting of SIT is extremely rare. To the best of our knowledge this is the first report of a uterus PEComa patient with SIT. There is no evidence that the presence of SIT predisposes one to develop PEComa. The clinicians should keep in mind that uterus PEComa may accompany SIT.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.


PTEN gene mutation was detected in his serum. An mTOR inhibitor had been used to treat his lung adenocarcinoma, but he developed aspiration pneumonia and died of respiratory failure. Autopsy revealed that the lung adenocarcinoma had metastasized to cavernous hemangiomas of the right atrial appendage and liver, to cerebellar dysplastic gangliocytoma and to multiple organs such as the liver, kidney, adrenal glands and spine. This is the first reported case of Cowden’s disease with multiple tumor-to-tumor metastases.Tumor-to-tumor metastasis is a rare phenomenon, but it has been suggested to be more frequent in patients with hereditary cancer syndrome. We report an autopsy case of tumor-to-tumor metastasis in a 75-year-old male. At 6 months before his death, the patient complained of hoarseness and dysphagia, and clinical whole-body examinations revealed advanced lung adenocarcinoma , multiple skin verrucas, gastrointestinal polyposis, goiters, and cerebellar dysplastic gangliocytoma (Lhermitte-Duclos disease), while  Tumor-to-tumor metastasis (TTM) is a term describing the presence of two histologically distinct tumors at one location, each with different morphologic and immunophenotypic features . The mosPTEN gene mutation, a point mutation (TGT to CGT) at exon 5 in codon 136, was detected in his serum. Finally, a definitive diagnosis of Cowden’s disease was made. Everolimus, an mTOR inhibitor, was used for 10 days to treat the lung adenocarcinoma but was stopped after aspiration pneumonia began to develop. After several days, he died of respiratory failure.A 75-year-old Japanese male complained of hoarseness and dysphagia. By various whole-body examinations, he was diagnosed with lung adenocarcinoma . Also found were a right cerebellar tumor Fig.  and b, dMacroscopic examination showed white solid nodules of 10–20 mm in diameter located in S8, S9, and S10 of the left lung and in S7 and S10 of the right lung Fig. . Well-deAll tissue samples were fixed in 10 % formalin and embedded in paraffin. Several 4-μm sections were cut from each paraffin block. Hematoxylin-eosin (H.E.) and immunohistochemical stains were performed. Microscopic examination of the H.E. sections revealed lung adenocarcinoma including solid nodules with signet-ring cells Fig.  and acinPTEN hamartoma tumor syndrome caused by PTEN gene mutation. It is an autosomal-dominant disease characterized by the development of multiple hamartomas in the skin, mucosa, digestive tract, breast, and central nervous system, and presents a high risk for malignant tumors in some organs. The pathological findings of the present case fully satisfied the criteria for Cowden’s disease [PTEN mutation [We presented an autopsy case of a patient with Cowden’s disease and multiple TTM, and these findings agreed with previous reports that TTM occurred more frequently in patients with hereditary cancer syndrome . To our  disease . Breast  disease . Althoug disease , the rev disease  includedmutation –19. Moremutation . Thus, wmutation . Howevermutation , which iTo our knowledge, this is the first reported case of Cowden’s disease with multiple TTM from a lung cancer to cavernous hemangiomas and cerebellar dysplastic gangliocytoma. TTM has been considered a rare phenomenon even though it is a hereditary cancer syndrome. The results of the present autopsy case suggest that more case of TTM in hereditary cancer syndrome might be revealed through careful macroscopic examination and sampling of tumors at autopsy, followed by attentive microscopic examination.Written informed consent was obtained from the patient’s family for the 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.


High-grade gliomas of the spinal cord represent a rare entity in children. Their biology, behavior, and controversial treatment options have been discussed in a few pediatric cases. These tumors are associated with severe disability and poor prognosis. We report a case of a 4-year-old child diagnosed with an isolated glioblastoma multiforme of the conus medullaris. The patient underwent subtotal surgical excision, followed by adjuvant radiotherapy and oral chemotherapy. He is alive with mild neurologic deficits at 52 months after diagnosis. We describe the peculiar characteristics of this rare condition in pediatric oncology. We also provide an overview of current multidisciplinary therapeutic approaches and prognostic factors for this disease. Primary spinal cord tumors are rare in children and account for less than 1% of all central nervous system (CNS) cancers .Low-grade astrocytomas represent the most frequently found histological type of intramedullary lesion (50%–88% of cases), approximately 1%–3% are high-grade gliomas, whereas non-glial tumors are less common [In this paper, we report a rare case of spinal GBM of the conus medullaris that occurred in a 4-year-old child. We further describe the peculiar characteristics of this rare entity in childhood and illustrate the current multidisciplinary therapeutic approaches.A 4-year-old boy reported a 6-month history of night-time worsening pain of both of the lower limbs and a subsequent appearance of urinary retention and weight loss. Neurologic examination demonstrated right leg paresis with bilateral hyporeflexia of the lower limbs. There were no neurologic abnormalities of the upper limbs and cranial nerves. No saddle anesthesia was observed.Brain and spine magnetic resonance imaging (MRI) revealed a large heterogeneous ovoid mass of 8 cm in the major diameter that filled the spinal canal between T11 and L3 , lysine 27 on histone H3 (H3K27), and isocitrate dehydrogenase 1 (IDH1) genes were performed, but no overexpression or mutations were identified.Mutational analyses for tumor protein 53  [The child underwent focal radiotherapy of the conus medullaris lesion (a dose of 4500 cGy delivered in 25 fractions) with temozolomide (TMZ, with a daily dose of 75 mg/mThe patient underwent neuro-rehabilitation with a gradual improvement of his motor weakness. He was soon able to stand and walk with a walking device. At 52 months after surgery, his motor impairment was almost completely resolved, although the patient continued to have a mild urinary retention syndrome. Late follow-up imaging confirmed complete remission in this patient (Fig. Pediatric intramedullary GBM are uncommon clinical entities. The commonly reported locations of GBM are the cervicothoracic segments, with the cervical spine being the most affected region, followed by the thoracic spine . The isoClinical features depend on the region of the spinal cord involved and the growth rate of the tumor, irrespective of the histological subtype. The most common symptoms in children include pain, motor regression, gait abnormalities, torticollis, and progressive kyphoscoliosis.Based on imaging, the criteria for the differential diagnosis of these lesions in children have been determined . Our casSimilar to the treatment strategy for brain lesions, gross total resection of the spinal cord lesion, confirmed by early postoperative MRI, followed by adjuvant treatment consisting of radiotherapy and chemotherapy, has been recommended .Historically, pediatric GBM has been treated with adjuvant radiotherapy followed by cytotoxic drugs either as single agents or in combination. Unfortunately, none of the chemotherapeutic regimens has been demonstrated to be superior over the others .Due to the lack of alternative treatments with superior clinical efficacy, radiotherapy in combination with concomitant and adjuvant TMZ is widely used by pediatric neuro-oncologists based on the efficacy of this strategy in adults . This trBecause of the high propensity of spinal GBM to disseminate, whole spine irradiation has been proposed. However, the role of prophylactic craniospinal irradiation remains unclear . Taking Pediatric GBM usually has a grim prognosis. Survival ranges from 6 to 16 months, with a mean survival of 12 months after the diagnosis .The extent and location of the lesion as well as the feasibility of a gross total resection, especially in younger patients, are the factors that most affect a patient’s prognosis . DespiteEven after 52 months of follow-up, our patient is not considered to be cured. Intensive rehabilitation aiming at optimal clinical recovery has promoted a good quality of life and the ability to perform daily life activities without support.The good clinical outcome observed in our case might partially be due to the absence of known negative prognostic molecular markers described in pediatric high-grade gliomas such as TP53 and H3K27 , 15. The


Introduction. Actinomycosis can affect any organ of the body, although cutaneous fistulas are common in actinomycotic infections, and other organs such as the bladder are only rarely involved. Case Presentation. Herein we report and discuss a young male patient with primary vesical actinomycosis. A 23-year-old man was hospitalized complaining of intermittent gross hematuria over a 6-month duration. The patient underwent a cystoscopic examination under general anesthesia; an edematous, hyperemic, wide-based mass, which protruded from the dome of the bladder, was seen and incompletely resected. The histopathological examination of the material showed Actinomyces organisms surrounded by inflammation and a photomicrograph showed the microorganism. After confirmation of bladder actinomycosis, the patient received penicillin. A CT scan of the abdomen and pelvis showed no evidence of the mass at the postoperative 6th month. Cystoscopic examination showed complete healing of the transurethral resection area at the dome of the bladder. Conclusion. In conclusion, we believe that the gold standard treatment for vesical actinomycosis should include the combination of a transurethral resection of the mass and long-term penicillin treatment. Actinomyces israelii and was first described in 1878 by Israel; however, at least six species of Actinomyces are pathogenic to humans. Actinomyces israelii is gram-positive, filamentous anaerobic, or microaerophilic bacterium normally found in the mouth and colon, but it is an opportunistic pathogen causing disease only in previously damaged tissue [ Actinomyces species are pleomorphic and branching filamentous bacteria that are currently classified in the genus Actinomyces of the order Actinomycetales, although they were once thought to be fungi because of their branching filaments [Human actinomycosis is generally caused byilaments . ActinomActinomyces species are susceptible to several antimicrobials including penicillin G, chloramphenicol, tetracyclines, erythromycin, clindamycin, imipenem, streptomycin, and cephalosporins. Penicillin G is the main drug used regardless of the severity and location of actinomycosis. Fluoroquinolones, aztreonam, fosfomycin, and other aminoglycosides generally have poor activity against Actinomyces species [ species . For pen species . OrdinarHerein, we report and discuss the case of a young male patient with primary vesical actinomycosis in terms of history, symptomatology, laboratory and radiological findings, treatment modalities, and follow-up.A 23-year-old man was hospitalized complaining of intermittent gross hematuria over a 6-month duration. He had previously undergone a left nephrectomy due to hydronephrotic atrophy. Additionally, he had undergone an unsuccessful cystoscopy and open bladder surgical attempt due to scar formation in the perivesical area in another hospital. The physical examination was unremarkable and urine analysis showed abundant erythrocytes without leukocytes. The urine culture was sterile and urine cytology showed no evidence of malignant cells. Intravenous urography showed a filling defect on the dome of the bladder. A computed tomography (CT) scan of the abdomen and pelvis showed a mass lesion of about 5 × 4 × 3 cm in size, involving the urinary bladder dome and invading the rectus fascia and muscles . Actinomyces microorganisms surrounded by inflammation (H&E, 200x, The patient underwent a cystoscopic examination under general anesthesia; an edematous, hyperemic, wide-based mass, which protruded from the dome of the bladder, was seen and incompletely resected. The histopathological examination of the material showedAfter confirmation of bladder actinomycosis, the patient received treatment as follows: penicillin at 20 million U/day intravenously for 2 weeks and then 6 million U/day intramuscularly for 4 weeks, followed by ampicillin at 1,500 mg/day orally for a total period of 4 months. A CT scan of the abdomen and pelvis showed no evidence of the mass 6 months postoperatively . CystoscThe incidence of genitourinary actinomycosis is very low. However, actinomycosis of the kidney, bladder, and scrotum has been previously reported –7. PelviVesical actinomycosis usually occurs via direct extension from primary infection of the pelvic organs. Imaging of abdominal and pelvic actinomycosis does not typically help with diagnosis since the disease usually presents as an abscess or a mass lesion and is commonly mistaken for a tumor. Diagnosis of vesical actinomycosis is often delayed due to the possibility of an urothelial malignancy carrying greater indices; initial diagnosis is often difficult and it is usually misdiagnosed as an urothelial or urachal tumor. In our case, the CT indicated a bladder tumor. In vesical actinomycosis, cystoscopy and endoscopic biopsy are recommended. We believe that an endoscopic biopsy is superior to a needle biopsy since it can be carried out under direct visualization. However, a biopsy does not allow the confirmation of radiologic diagnosis in other organs such as the kidney. Actinomycosis can cause hydronephrosis. Many hydronephrosis cases due to actinomycosis infections were reported in the literature , 12. In Primary bladder actinomycosis can be misdiagnosed as a bladder tumor and may be treated by partial or radical cystectomy. Lim et al. performed partial cystectomy in a patient with urachal actinomycosis since they could not rule out malignancy despite the use of preoperative PET and CT ; they reThe contribution of urine analyses is limited in the diagnosis of vesical actinomycosis. Pyuria and microscopic hematuria are found in the urine analysis and urine culture is usually sterile; there are only a few reported cases diagnosed by urine analyses , 17. JanSurgery is indicated for the resection of necrotic tissue, excision of sinus tracts, and drainage of empyemas or abscesses. It is believed that surgical resection reduces the total treatment period to 3–6 months . AlthougPreoperative diagnosis of primary bladder actinomycosis is difficult due to the nature of its clinical presentation, and it can be misdiagnosed as a bladder tumor or treated as bladder cancer. In many instances, it is misdiagnosed as a vesical or urachal tumor and is usually diagnosed postoperatively through pathological confirmation. The true diagnosis of actinomycosis may be missed because of the rarity of this condition. Therefore, actinomycosis cases should be reported in order to increase the awareness of this disease.


Metastasis of a primary osteosarcoma to the muscles is extremely rare. As there have been few reported cases, the necessity of surgical treatment for such metastatic lesions remains controversial. We present the case of a primary osteosarcoma with development of a solitary metastasis to the trapezius muscle during chemotherapy for pulmonary metastasis. The patient was a 51-year-old man diagnosed with osteosarcoma of the right tibia. After undergoing chemotherapy and femoral amputation, he developed pulmonary metastasis. Chemotherapy was reinitiated, however, after approximately 1 year a palpable tumor was identified in the patient’s right shoulder. This tumor grew and was associated with pain in the right shoulder. It was surgically removed 3 years after the re-initiation of chemotherapy. The pathological diagnosis was osteosarcoma with metastasis to the trapezius muscle. Although the patient died of respiratory failure due to pulmonary metastasis 14 months after resection of the metastatic lesion in the trapezius muscle, no new extrapulmonary metastasis was observed after the resection. The metastasis of osteosarcoma is mainly hematogenous and it has been demonstrated that, at the time of diagnosis, approximately 80% of osteosarcomas have already metastasized if micrometastases are included . The vasThe patient, a 51-year-old man, consulted a local hospital after noticing idiopathic pain in his lower right thigh. The patient had no medical history of note. A radiography revealed osteolysis with partial ossification  and doxorubicin (30 mg/m2 on days 1 to 2) was not effective and the tumor continued to grow. On completion of two courses of chemotherapy a pathological fracture occurred and an amputation of the right femur was performed. The histological response rate of the resected specimen was less than 10% of the entire lesion. Following surgery, five courses of adjuvant chemotherapy with ifosfamide (3 g/m2 on days 1 to 4) and VP-16 (75 mg/m2 on days 1 to 4) were administered and the patient was discharged as disease free.During follow-up, a computed tomography (CT) scan at 9 months after discharge revealed multiple metastases in both lungs (Figure At this time no apparent metastatic lesions were observed. The administration of preoperative neoadjuvant chemotherapy with cisplatin (120 mg/met al.[It has been demonstrated that the metastasis of malignant tumors to skeletal muscle is extremely rare, with an incidence of approximately 1% ,3. Accoret al., musculaet al.,6, with Previous reports have demonstrated that the incidence of soft tissue metastasis due to osteosarcoma is approximately 2% -11. Howeet al.[At present, conservative treatment with the systemic administration of anticancer agents along with radiation therapy is often chosen to treat metastatic lesions, and the necessity of surgical treatment remains controversial. Yamada et al. reportedOur patient received systemic chemotherapy for both pulmonary and muscle metastases. Despite achieving good control of the pulmonary metastases, we were unable to control the metastatic lesion in the muscle with chemotherapy. The resected muscle lesion revealed living cells in the majority of the tissue. A previous study demonstrated differing sensitivity patterns to cytostatic agents in cell culture studies of metastatic pulmonary and muscular osteosarcoma, suggesting that therapy may have resulted in positive selection for a tumor clone which is resistant to therapy and/or more metastatic than other subpopulations within the tumor . AlthougWe have presented an extremely rare case of primary osteosarcoma with solitary metastasis to the trapezius muscle that developed during chemotherapy for pulmonary metastasis.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.CT: Computational tomography; H & E: Hematoxylin and eosin stain; MRI: Magnetic resonance imaging; PET-CT: Positron emission tomography-CT; SUV: Standardized uptake value.The authors declare that they have no competing interests.SY, SN, HS, and MY managed the case and drafted the manuscript. SN, TS, IK, and YI collected the findings and participated in the design of the study. AT performed pathological examination and figure preparation. SK was involved in the design and coordination of the study and helped to draft the manuscript. All authors have read and approved the final manuscript.


Esophageal leiomyomas (ELMs) are rare but described in the literature. They are usually benign and do not require resection unless they are large and symptomatic. Most of such masses arise from the muscularis mucosa. It is very uncommon to find epithelial dysplasia overlying a subepithelial leiomyoma. A review of the literature reveals only one prior case of ELM with an overlying epithelia dysplasia and here we report a second case. Leiomyoma (LM) is the most common benign esophageal tumor . It is u in situ  junction, found on surveillance EGD Figures . GE junc Figures . Histopa Figures . Immunoh Figures . A diagn Figures . No procELM was first well described by von Rahden et al. in 2004 . AlthougEGD, EUS, CT of chest, and so forth may aid in diagnosis. On endoscopy, LM is identified as a submucosal, freely movable mass with intact mucosa . EUS finTreatment for ELMs depends on multiple factors, including tumor size, location, appearance, and the patient's symptoms and overall conditions , 16. AsyThe coexistence of an epithelial lesion and a subepithelial lesion is rare. To our knowledge, twelve patients in ten case reports with carcinoma located in the mucosa overlying a benign tumor have been reported –21. All In conclusion, leiomyomas of the esophagus can rarely be found underneath a severely dysplastic mucosa. Endoscopic removal is a suitable option for lesions arising from muscularis mucosa, if detected early.


Renal cell carcinoma comprises about 2% of adult tumors. The overall 10-year survival rate of patients with RCC after nephrectomy is about 18-27%. The incidence of metastasis of initial RCC is about 24-28%, but this rate after nephrectomy is as high as 51%. The most common site of recurrence is the lung, however liver and bone metastases are common. There are many reported cases with late metastasis, however isolated late metastasis in the gastrointestinal tract especially duodenum is very rare.Herein we report our experience with a case of gastrointestinal bleeding secondary to metastatic renal cell carcinoma to duodenum, 16 years after nephrectomy.To the best of our knowledge, about 30 of such cases have been reported in the English literature. Many of the previous cases have been part of disseminated disease and isolated duodenal metastasis is very rare. The longest reported duration between nephrectomy for renal cell carcinoma and duodenal metastasis has been 13 years, thus it seems our case to be also unique because of very late duodenal metastasis.  Herein we report our experience with a 61-year-old man presented with melena 16 years after nephrectomy that was diagnosed to be metastatic RCC. The patient underwent Whipple’s operation with excellent postoperative course. A 61-year-old man was presented with melena. He was in a good health condition. His past medical history showed right kidney nephrectomy (16 years ago) with the diagnosis of clear cell RCC without any further treatment. He had been completely normal during the last 16 years.Laboratory findings were: Liver function tests were normal: ALT=19 U/L , AST=25 U/L , Alkaline phosphatase=193 U/L 6/µl, and Platelet: 263000/µlComplete Blood Count was normal: WBC: 6000/µl, RBC: 5×10Physical examination showed normal heart and lung examination. There was no lymphadenopathy. Blood pressure, pulse rate, heart rate, and temperature were all unremarkable.Stool occult blood test was positive in several occasions, so the patient underwent upper and lower gastrointestinal endoscopy. An ulcer was found in the second part of duodenum with fine oozing of blood . ImmunohWith the pathologic diagnosis of metastatic RCC, abdominal CT scan was performed that showed a mass in the distal pancreas measuring 7 cm in greatest diameter with central necrosis, most probably arisen from duodenum extending to the pancreas . WhippleThis case is being reported by the informed consent of the patient. 4 Most common sites of metastatic RCC are the liver and lung.2 Metastases to the pancreas and small intestine are rare, but can present as gastrointestinal bleeding.5 About 4% of RCCs metastasize to the small intestine, of these; the duodenum is the least frequent site and can be involved by direct invasion of the tumor, or through lymphatic, transcoelomic or haematogenous spread and also direct invasion of pancreatic metastasis.5 In our patient, at the time of surgery, the main bulk of tumor was in the duodenum with extension to the head of pancreas.The behavior of RCC is completely unpredictable. Metastatic masses can be the first presentation of RCC or can be diagnosed years after the initial diagnosis and nephrectomy.6 Primary presentation of metastatic RCC as gastrointestinal bleeding has been rarely reported.7According to the previous reports, duodenal involvement of RCC can be presented with jaundice, anemia and gastrointestinal bleeding, malabsorption and obstruction.8The longest duration between nephrectomy and duodenal metastasis was 13 years in previous reports.9Diagnosis of duodenal metastasis in RCC most commonly has been made by endoscopy. On endoscopy, the lesion can be seen as an ulcer, submucosal mass with ulceration or multiple nodules or small polyps of varying sizes.10Some patients present with concomitant metastasis in more body locations or other segments of the intestine such as the colon and duodenum.8Our case presented with melena and duodenal ulcer 16 years after right nephrectomy for RCC. In previous studies, most of the duodenal metastasis of RCC was after right kidney nephrectomy.9Most of the metastatic RCCs in the duodenum were primarily in the second portion with and without pancreatic involvement.2 In previous reports, most of the patients with duodenal metastasis of RCC were treated with Whipple’s operation; however, there were successful surgeries of duodenal saving segmental or wedge resection.3,11 Any type of metastasectomies can increase the survival of the patient.5The treatment of choice for localized metastatic RCC is surgery.To the best of our knowledge, less than 30 cases of RCC with duodenal metastasis have been reported in the English literature . The lonAs a conclusion, distant metastasis of RCC can be presented very late with unusual and unpredictable symptoms. In all patients with a history of RCC, GI bleeding should be considered as a possible cause of metastasis.


Congenital teratoma is a rare condition and is a germ cell tumor composed of elements from one or more of the embryonic germ layers and contain tissues usually foreign to the anatomic site of origin. We report a case of a neck tumor diagnosed during pregnancy, initially thought to be a goiter. After birth the neck mass kept growing until it compressed the trachea and produced respiratory failure. The infant had a difficult tracheal intubation because of the compressing mass. The staff decided to surgically remove the neck mass. After that, the infant became eupneic. The histological analysis showed a mature teratoma with no atypias. We report a case of an infant with a neck mass diagnosed by ultrasound during pregnancy which was initially supposed to be a congenital goiter. Two doses of levothyroxine were administered into the amniotic fluid. The goiter kept growing after birth until it caused severe respiratory distress by compressing the trachea, necessitating immediate tracheal intubation. The tumor was surgically resected and the patient went eupneic for the first time in his life. The histological analysis demonstrated a mature teratoma with no atypias. Thyroid hormone substitute therapy was started and the infant is thriving well.Congenital thyroid teratoma is a rare conditionA 2-months-and-20-days-old Brazilian white male infant weighing 4.2 kg was admitted to the pediatric intensive care unit of our hospital  in acute respiratory distress and was immediately intubated and placed in mechanical ventilation.From a routine ultrasound during pregnancy, the fetus had been diagnosed with a cervical mass, considered initially to be a goiter  by doctoAfter hospital discharge, he was observed by a pediatric endocrinologist who started research on thyroid disorders. Meanwhile, the infant maintained a euthyroid state, receiving no treatment, waiting for more investigation on the cause of the neck mass. However, the cervical mass kept visibly growing, was palpable and the infant presented a laryngeal stridor that was still attributed, by the pediatrician who followed the infant, to laryngomalacia. In the few days preceding hospitalization at our institution, the infant became increasingly dispneic each day, as related by his mother. One day, after choking and vomiting during breastfeeding he became hypotonic and went into acute respiratory distress.He was admitted to our pediatric intensive care unit 25 days after he had been discharged from the other hospital, and was immediately intubated. An X-ray showed a small amount of interstitial infiltrate, compatible with aspiration pneumonia. However, the respiratory distress was attributed mainly to an upper airway obstruction. It was difficult to tracheally intubate the infant; only an uncuffed 2.5 mm endotracheal tube (ETT) was able to be inserted into the trachea and it was difficult to place this in the right position. The X-ray after intubation showed the ETT in a high position and the trachea displaced to the right . MagnetiMeanwhile, we started investigation into the cause of the neck mass and performed blood tests on the infant: thyroid hormones were in the normal range (free thyroxine (T4): 1.3 ng/dL and thyroid-stimulating hormone (TSH): 4.7 ng/dL). Calcitonin levels, for investigations into potential malignance, were normal , as was the alpha-fetoprotein: 505 µg/L.We decided to remove the cervical mass, since it was causing the tracheal obstruction. The surgery lasted 35 minutes and was uneventful. The mass was well circumscribed and could be easily dissected, weighed 20 grams and measured 33×61×45 mmLevothyroxine was started  as thyroid hormone substitute therapy and the infant is thriving well according to the pediatric endocrinologist that continues following the patient.3. They are most common during early childhood and the most common location is the sacrococcygeal region in children and the gonadal region in adults4. The frequency of these embryonic tumours is about 1:20,000–40,000 live births. However, only 1.5% to 5.5% of all pediatric teratomas are placed in the neck region. These tumours are usually solitary, with no other associated congenital malformations or chromosomal abnormalities4. Although 95% of all teratomas are benign, the cervical teratomas if not properly treated, lead to death in 80% of the cases due to obstructive respiratory distress5.Teratomas originate from multipotent primitive germ cells and result in different tissues, diverging from the anatomical site of originIn this case, the acute clinical presentation of the neck mass with severe respiratory distress, needing ready intervention and immediate tracheal intubation should alert all pediatricians to the risk of these neck masses, and consider it as a potentially fatal case. In the presented case, surgical removal of the neck mass was both diagnostic and therapeutic.5.Thyroid teratoma is rare in infants, it is usually benign, and can cause airway compression depending on the site and size of the mass. The likelihood of a malignant thyroid teratoma is low in infants, however it could be fatal by causing upper airway obstruction. Therefore, surgical resection is required both for diagnosis and treatment. If the surgical removal is a success, the long-term outcome and quality of life should be goodWritten informed consent was obtained from parents of the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor of this journal. The authors describe an interesting case, with a positive outcome despite some delay in diagnosis.The title and the description of the case are all well written.discussion and the conclusion require more work. This case brings up an important and not so rare clinical scenario in Paediatric Critical Care, i.e. the difficulties inherent to extrinsic airway compression by a tumour. The authors, on their discussion, should take advantage of this case and discuss the approach to airway extrinsic compression, the use of extra-corporeal life support (ECLS) as a back-up plan, who should be present in such situations , etc… The clinician could barely secure the child’s airway, and the child survived and was able to have the teratoma resected. But it could have been a disastrous outcome. There are protocols for such scenarios, and they should be mentioned and described for the reader to be prepared when facing with similar situationsHowever, theI have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above. et al described in a case report of severe acute respiratory distress by tracheal obstruction due to congenital thyroid teratoma. This case report adds knowledge to the literature, not only by tumor description, but also the clinical management of the case.ColettiTumor description is interesting, however I believe that the authors should also discuss in this article the clinical management of malacia in premature, since this is very common in neonatal clinic. I believe that this discussion would make the most interesting article and is also an opportunity to develop the discussion of the article, which I believe is short. For this reason there are some points which need to be considered and are necessary clarificationsThe patient was born with what gestational age?After birth was not performed control with imaging for the mass?What are the results of thyroid hormones tests after birth?The staff investigated other causes for laryngeal stridor before discharge?Suggestions:Add data:The discussion could  discuss the differential diagnosis of stridor. The staff not investigated a patient with stridor, attributing the malacia. Many patients in neonatology have stridor and I believe that this article could show that there is need for investigation in stridor, because sometimes the differential diagnosis can be serious pathologies.Case reports are interesting, but I believe it should have a good discussion of the various aspects involved in the pathology, in which case I would like that you develop the discussion and mainly develop the discussion about the differential diagnosis.We have read this submission. We believe that we have an appropriate level of expertise to confirm that it is of an acceptable scientific standard. I would like to thank Dr Martinez and colleague for the remarks and suggestions above.The patient was born with 31 weeks of gestational age, immediately after the second administration of levothyroxine in the amniotic fluid due to a chorioamnionitis.After birth new imaging control was done in the previous hospital and in our medical center (figure 3).After first hospital discharge, the patient was followed by a pediatric endocrinologist who asked for thyroid hormone tests which resulted normal for the age .Since the patient was born in other hospital we could not know if the staff did any other investigation for other causes of laryngeal stridor. In our medical center, the patient was admitted in a critical clinical status and the surgery was promptly performed.I hope I have clarified your doubts and concerns about this case report. Thank you for the review.Answering the questions regarding this case report:


The clinical case of a 65-year-old woman with an incidentally detected left-sided mass in the upper renal pole is presented. A functional adrenal tumor was excluded. The mass was removed retroperitoneoscopically. The perioperative period was uneventful. The histopathological examination revealed a heterotopic intrarenal adrenocortical oncocytoma.Adrenal oncocytic neoplasms are very rare, with, to the authors’ knowledge, only 159 described cases so far. Most cases are non-functioning adenomas that can reach a considerable size. Only 10 heterotopic adrenal oncocytomas have been described . Although the intrarenal adrenal rest is the most frequently appearing variant of adrenal heterotopia, to the best of our knowledge, this report is the first description of an intrarenally growing adrenocortical oncocytic adenoma.In addition to retroperitoneally located oncocytomas, this case could be interesting for urological practice because there are no diagnostic features which could provide a secure preoperative diagnosis of an adrenal oncocytic neoplasm and its malignant variant. Generally accepted indications for surgery of adrenal masses have to be respected. The definitive pathologic diagnosis is in most cases surprising because of its rarity. Benign adrenal oncocytic neoplasms do not require any adjuvant treatment. The oncocytic variant of adrenocortical carcinoma generally has a poor prognosis. A 65-year-old Caucasian woman with a history of arterial hypertension underwent an ultrasound examination of the kidneys performed by her general practitioner in 2012. A left-sided suprarenal mass measuring about 5 cm was detected. The physical examination showed no palpable masses in the abdomen, and the peripheral lymph nodes were not enlarged. A 3-T magnetic resonance imaging (MRI) of the adrenal glands presented a heterogeneously enhancing mass measuring 48×48×33 mm in the left apical renal pole in contact with the adrenal gland . The finen bloc with the mass  was uneventful. No additional treatment was necessary.17. Most of the papers describe only single patient case reports.Oncocytic neoplasms of the adrenal glands are extremely rare. A PUBMED search up to December 2013 uncovered only 159 cases of adrenocortical oncocytomas since the first description of this tumor in adrenal in 19862 and rarely in the respiratory tract4, choroid plexus5 and adrenal glands. Oncocytic neoplasms of the adrenal glands, with, to the authors’ knowledge, only 159 previously described cases, are an extremely rare phenomenon. Most of these tumors were discovered as incidental findings on CT or MRI as non-functional adrenal masses. Various malignant potential can be determined in 20% of adrenocortical oncocytic neoplasms6. Oncocytomas arising from the heterotopic adrenocortical tissue have been described in only 10 case reports, seven cases were located intraspinally13 and three cases in the retroperitoneum16. An oncocytic adenoma arising from heterotopic intrarenal adrenocortical tissue was not suspected in the preoperative assessment of this case because this entity has not been described previously. However, in addition to retroperitoneally located oncocytomas, this should be considered in the future as a differential diagnosis in cases presenting with an intrarenal or retroperitoneal mass.Oncocytic neoplasms or oncocytomas are mostly benign tumors which commonly occur in the kidneys, thyroid, parathyroid, pituitary and salivary glands18.This paper contains the first report on an oncocytic adenoma arising from the heterotopic intrarenal located adrenal tissue. Heterotopic adrenal tissue or adrenal rest presented mostly by cortical structures is more frequently located in the kidney. Other sites such as the celiac trunk, epididymis, spermatic cord, ovary, broad ligament, liver capsule, gallbladder, pancreas and spleen are rare19, which are commonly used in the histological diagnosis of adrenocortical malignancies, are not applicable to adrenocortical oncocytic neoplasms because all tumors have eosinophilic tumor cytoplasm, diffuse architecture and nuclear atypia. The modified Lin-Weiss-Bisceglia system differentiates between major, minor and definitional criteria for malignancy6. None of these criteria is present in benign adrenal oncocytic neoplasms. Masses with uncertain malignant potential demonstrate the presence of one to four minor criteria  in absence of major criteria . In adrenal oncocytic carcinomas any of the major criteria could be present.Oncocytic neoplasms are mostly encapsulated masses with a brown or yellow surface on cut-section. The radial scar can be absent. Oncocytic neoplasms microscopically consist of so called oncocytes, large cells with rich eosinophilic granulations due to the high concentration of mitochondria. The Weiss criteria20. The bulk size cannot be used as a reliable criterion to estimate the risk of malignancy.There are no specific criteria on both computed tomography and MRI and MRI with chemical shift subtraction for adrenal oncocytic neoplasm and its malignant variant21. In rare cases, an adrenal oncocytic neoplasm can produce catecholamines, cortisol or testosterone21.Eighty-three percent of adrenal oncocytic neoplasms are non-functioning massesThe therapeutic standard is a minimally invasive adrenalectomy. In cases of a large mass, infiltration of surrounding structures, and lymph node bulks, an open surgery approach should be chosen.21. The oncocytic variant of adrenocortical carcinoma has a poor prognosis, with a postoperative recurrence rate of 75%, and a tumour-related mortality of 40% in a small group of 24 patients with a median follow-up of 21 months (range: 1 – 180 months)21. An adjuvant or palliative chemotherapy with mitotane can be administered in patients with adrenocortical carcinomas with beneficial effects22, but there is no evidence of efficacy when applied to oncocytic variant.Today, there are no recommendations for the follow-up of benign adrenal oncocytic neoplasms. Only one local recurrence of a neoplasm that was originally diagnosed as benign has been described and fulfilled the criteria of uncertain malignant potentialTo the authors’ knowledge, this case report presents the first description of a heterotopic intrarenally located adrenocortical oncocytoma. Although being a rare location, this case is worth mentioning, given the challenging situation with regard to the diagnostics and differentiation from the potentially aggressive malignant lesions. Taking in account the absence of radiological criteria of a benign character, this tumor should be considered and treated as a malignant lesion, although a minimally invasive approach should be chosen when possible. The definitive pathologic diagnosis is in most cases surprising because of the rarity of this type of neoplasm and radiological appearance mimicking renal cell carcinoma or adrenal carcinoma. Benign adrenal oncocytic neoplasms do not require any adjuvant treatment.Written informed consent for the publication of clinical details and clinical images was obtained from the patient. This is a report of a very interesting case of a heterotopic intrarenal adrenocortical oncocytoma.As the authors stated this may be the first reported case in the literature.The major concern is the appearance of the mass in relation to the kidney at figure 1, exactly at the location of left adrenal gland. The authors stated that they performed a retroperitoneoscopical enucleation and, intraoperatively, the mass had a very thin, poorly defined pseudo-capsule which was adhering to the renal parenchyma as well as to the macroscopically inconspicuous adrenal gland.These observations raise the possibility that this mass was in fact arising from the adrenal gland itself and not from a heterotopic location within the kidney. A tumor of 4.5 cm located intrarenally would be difficult to enucleate without interfering with any renal parenchyma and therefore vascular structures - causing bleeding.The provided histopathological figure displays only diffuse sheets of oncocytic cells without any evidence any surrounding renal tissue.I would recommend a repeat discussion with the pathologist to see if this not really an adrenal oncocytoma, which would be still a rare entity, albeit with more than 50 cases reported in the literature.I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above. Dear Professor Turkeri,Thank you for your evaluation.Indeed, this tumor arose from the renal parenchyma and in fact left adrenal was intact and had no continuous contact to the tumor.We have discussed your concerns with the pathologist and in the revised version of the article we will provide a pathology slice, where the nearby renal parenchyma and normal adrenal cells could be seen direct by the tumor, making the final diagnosis undoubtful.With best wishes,Konstantin Goding, Nicole Bang and Yuri Tolkach The authors report a heterotopic adrenal oncocytoma in a 65 year-old lady with essential hypertension. A routine ultrasound detected a 5 cm adrenal mass, however, MRI showed a 4.8 x 4.8 x 3.3 cm mixed intensity upper polar renal mass. Features were not suggestive of a malignant tumour. Urinary and blood work up excluded a functioning adrenal adenoma. The oncocytoma was removed retroperitoneoscopically with the zero-ischemia technique.The sequence of the work up is not clear: was the MRI done first; or were urinary catecholamines and metanephrines, aldosterone-renin ratio and serum cortisol determined first? In the latter case, why was a pheochromocytoma suspected when neither the clinical manifestations nor imaging (MR) suggested a pheochromocytoma?The authors have rightly noted that the imaging characteristics of benign and malignant oncocytoma are not clearly described in the current literature - they were entertaining a diagnosis of an RCC pre-operative. Did the authors consider doing a biopsy prior to surgical intervention?  It would be pertinent to mention indications for biopsy in such a situation, and discuss its merits and demerits. It is a well written report, however, I have the following observations on the current submission:I have read this submission. I believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above. Dear Dr. Ather,Thank you for you valuable comments.With regard to the first point: hormonal evaluation was done after MRI investigation to exclude the probability of hormone-producing tumor, while the tumor was located very close to the adrenal, though the probability was indeed very small. It was done according to the local guidelines.Concerning the second point, based on contemporary evidence it is hard to discuss the necessity of biopsy both in adrenal and renal tumors. We did not consider this diagnostic modality. In our opinion, in this case there was no place for the biopsy.With kind regards,Konstantin Godin, Nicole Bang and Yuri Tolkach


We report a case of adenocarcinoma arising from a sacrococcygeal mature teratoma in an adult female. A 62-year-old female was diagnosed with a presacral tumor 10 years ago. Pelvic computed tomography (CT) demonstrated a presacral heterogeneous tumor, containing multiloculated cystic area and enhanced solid component with calcification. Percutaneous needle biopsy for the solid component of the tumor identified an adenocarcinoma and the patient was diagnosed as having a sacrococcygeal teratoma with malignant transformation. Abdomino-sacral rectal resection with sacral amputation at the upper edge of the S5 was performed. The pathological diagnosis was adenocarcinoma derived from a mature teratoma. The tumor cells had infiltrated the rectal wall. After 7 months, a follow-up CT demonstrated swelling of the right inguinal lymph nodes and a right inguinal lymphadenectomy was performed. Pathological examination showed metastatic lymph nodes. The patient is doing well 21 months after the second surgery, with no signs of recurrence. Teratomas are a type of germ cell tumor, which are mostly benign, and dominative in adult females , 2. The Although most of the patients with mature teratoma undergo surgical resection due to concern about the potential for malignant transformation, others choose to be followed-up without resection. However, early detection of malignant transformation is often difficult even with current imaging modalities, and it is unclear when the malignant transformation occurs during the follow-up period. We report a case of adenocarcinoma arising from a sacrococcygeal mature teratoma in an adult female who had been followed-up for 10 years.A 62-year-old female who presented with anal pain for 3 months was referred to our hospital. Ten years at another previous hospital she was diagnosed as having both myoma of the uterus and a presacral tumor on computed tomography (CT). Five years later, a repeat pelvic CT scan showed no change in shape or size of the presacral tumor Figure . On admiAbdomino-sacral rectal resection with permanent colostomy was performed. On laparotomy, there was no evidence of liver metastasis or peritoneal dissemination. After the large uterine myoma was raised, the tumor was palpable on the right side of the rectum. The uterine myoma was not removed. The rectum was fully mobilized and the periosteum of the upper edge of the S5 was exposed. The second step was sacral resection. The patient was placed in a jackknife position, a median incision including the anus was made, and a sacral amputation was carried out using the chisel and hammer at the upper edge of the S5. Following this, the tumor was completely resected from the pelvic floor Figure .Macroscopically, the tumor was a multilocular cystic lesion containing sebaceous matter, hair, and follicles Figure A. The paAfter 7 months, a follow-up CT demonstrated swelling of the right inguinal lymph nodes Figure A. The adIt is generally accepted that the patients with sacrococcygeal mature teratomas are suitable for surgical treatment because of a malignant potential. Once the malignant transformation arises, only en-bloc radical resection offers the possibility of a good outcome. However, it is often difficult to achieve R0 resection when the tumor has become large or has invaded surrounding organs in the narrow pelvis. Therefore, various surgical approaches have been reported, including trans-abdominal, trans-sacral, and a combined abdomino-sacral approach –11. In oUnfortunately, the radial margin was microscopically positive (R1) in the present case. Although adjuvant chemotherapy, radiotherapy, and surgical re-resection were all treatment options, we did not administer any additional therapy. Several studies have investigated the efficacy of adjuvant chemotherapy and have shown that systemic therapy was beneficial to few patients with malignant transformation from a teratoma. However, the majority of these results were related to the efficacy of adjuvant chemotherapy in squamous-cell carcinomas arising from ovarian teratoma , 13. WheThe pathway from the perineal cutaneous part of the anal canal and the vulva to superficial inguinal nodes is recognized as the “superficial perineal accessory pelvic pathway” . In caseIn the majority of cases, when a CT and/or MRI scan reveals a sacrococcygeal teratoma, surgical resection is immediately selected because of concerns over malignant transformation. However, it is still unclear when the malignant transformation happens in a benign tumor and how we should observe patients who elect not to have surgical resection. A unique feature of the present case is that the patient had been followed-up by CT examination for 10 years at a previous hospital. The tumor size and shape did not change in the first 5 years, however, in the second 5-year period, there was a 1.4-fold increase in the size of the tumor and the solid component invaded the rectal wall. Although, clearly, an early diagnosis of malignant transformation offers the best hope of a successful outcome, it is also the case that once malignant transformation occurs, localized involvement and metastases can develop rapidly in some cases . In caseAnother debatable point is whether preoperative needle biopsy is essential. Percutaneous needle biopsy is crucial for an accurate diagnosis and it might allow for a consensus decision in the subsequent treatment of patients with germ cell tumors . HoweverIn conclusion, we describe an extremely rare case of a patient with a sacrococcygeal mature teratoma with malignant transformation into an adenocarcinoma. Surgical resection was effective for both primary and metastatic inguinal node disease. In case of patients with mature teratoma who opt for non-surgical treatment, fairly frequent follow-up is recommended.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


A few cases of a gastrointestinal stromal tumor (GIST) of the small intestine presenting as rupture have been reported in the medical literature. We report an unusual case of a large GIST of the jejunum that presented as a spontaneous rupture. A 70-year-old man was referred to our hospital because of fever and abdominal pain. An abdominal enhanced computed tomography (CT) scan detected a 10-cm tumor with heterogeneous staining, suggesting necrosis or abscess inside the tumor. The patient was treated with antibiotics but inflammation persisted and an operation was performed. Intraoperative findings showed an outgrowing 10-cm mass in the jejunum near Treitz's ligament. The tumor had ruptured with peritoneal metastasis. The solid parenchyma contained a focal area of necrosis within and the small ulcer located in the wall of the jejunum presented a communication with the large tumor cavity. H&E staining showed spindle-shaped cell proliferation, and immunohistochemical staining showed results positive for c-kit and CD34. The patient received a diagnosis of a GIST (high-risk group) of the jejunum and was treated with imatinib mesylate. The patient has remained in good health without recurrence or metastasis one year after the surgical procedure. Gastrointestinal stromal tumors (GISTs) are the most common mesenchymal tumor of the gastrointestinal tract . Most GI3, and C-reactive protein of 19 mg/dL. An abdominal enhanced computed tomography (CT) scan revealed a 10 × 10-cm solid tumor with a low-density area within, suggesting necrosis or abscess; additionally, a liver metastasis was suspected (Figure A 70-year-old man presented with symptoms of fever and abdominal pain. There were no episodes of gastrointestinal bleeding. The blood pressure was 117/86 mmHg, the pulse 80 beats per minute, and the temperature 38.2°C. Abdominal examination revealed tenderness and muscular defense in the left upper quadrant, and rectal examination revealed an empty ampulla. Blood tests showed a hemoglobin level of 7.2 g/dL, white blood cell count of 25,490/mmet al. and is widely accepted and used today [GISTs are visceral tumors arising from any site of the gastrointestinal tract. Approximately 60 to 70% of cases occur in the stomach, 25 to 35% in the small intestine, and 10% in the jejunum, whereas the esophagus, colon, rectum, and appendix, are rarely affected . Bleedined today -19 (Tabled today . The proed today .The introduction of imatinib mesylate, a tyrosine kinase inhibitor targeting KIT has provided a much needed chemotherapeutic option for patients with both resectable and unresectable GISTs. Despite the noted success of imatinib, surgical resection is the main treatment modality for primary GIST of any localization ,23. ImatIn conclusion, spontaneous rupture is a rare presentation of GIST and preoperative diagnosis is difficult because of the absence of pathognomonic signs or symptoms. The diagnosis should be suspected whenever there is a presentation of fever and abdominal pain in patients with an intraabdominal mass. In this case, GIST originating in the jejunum led to central necrosis and spontaneous rupture in the peritoneal cavity; however, local excision of the tumor associated with adjuvant therapy with imatinib mesylate was effective and these treatments remain the main modality of treatment of high-risk GISTs.Written informed consent was obtained from the patient for publication of this report and any accompanying images.CT: computed tomography; GIST: gastrointestinal stromal tumor; H&E: hematoxylin and eosin.The authors declare that they have no compating interests.SM was responsible for the writing. MT, HS, YK and HT participated in the clinical management of the patient. HO carried out the pathological examination. All authors read and approved the final manuscript.


Histoplasma capsulatum infection.Mesenteric cysts are uncommon entities and chyle- (lymph-) containing cysts are the rarest of this group. This is a case report of a 4-year-old boy with a mesenteric chylous cyst who was later found to have Chylous cysts are benign proliferations of lymph vessels which result from an obstruction in the lymphatic system. They account for approximately 3% to 9.2% of all paediatric lymphangiomas, although their incidence is unknown , 2.These lesions can present with symptoms such as abdominal pain, nausea, vomiting, anorexia, and changes in bowel habits; however, most commonly they are asymptomatic and are detected incidentally on physical exam or imaging. Although most mesenteric cysts are benign, these lesions occasionally cause complications, including intestinal obstruction, volvulus, or even torsion , 4.A 4-year-old boy presented to our hospital with a 6-month history of on and off vague abdominal pain and anorexia. Two weeks prior to admission, the pain remained vague but was associated with occasional nonprojectile vomiting and nausea; he however continued to have normal bowel habits. A day prior to coming to the hospital, he started having projectile Bilious Vomiting.He was admitted as a patient of acute absolute intestinal obstruction, possibly due to intussusception. The preliminary blood work revealed no derangement in function; that is, he had normal complete blood count, liver function tests, and renal function tests. Abdominal ultrasound was done, where reduced bowel peristalsis was seen, with no other remarkable findings. The plain abdominal X-ray showed a possible gross dilation of colon in the hepatic flexure with few air-fluid levels.On Exploratory Laparotomy, we found a very large (15 cm in diameter) mesenteric cyst within the small bowel mesentery located 30 cm from the ileocecal junction and obstructing a portion (10 cm) of small bowel lying above it Figures , and 3.Excision of the cyst together with resection and anastomosis of the affected bowl were carried out. We drained 750 mL of chylous fluid from the excised cyst.Histopathological findings  showed the presence of histoplasmosis spores within the lining of the cyst Figures , and 6.The patient had an uneventful postoperative recovery period. He was discharged 5 days later after full recovery. Parents report that he has since gained an appetite for food. He also is currently on itraconazole treatment.Chylous cysts are rare variants of mesenteric lesions, making up 7.3% to 9.5% of all abdominal cysts . There aAccording to Beahrs et al. , based o infective, type 2, chylous cyst.Furthermore, based on the contents of the cyst, the mesenteric cysts can also be classified into serous, chylous, hemorrhagic, and chylolymphatic cysts. The chylolymphatic cysts, as indicated by the name, contain both chyle and lymph. Therefore, in this patient, the cyst can be classified as anThese cysts can present with symptoms such as abdominal pain, nausea, vomiting, anorexia, and a change in bowel habits; however, most commonly they are asymptomatic and are detected incidentally on physical exam or imaging. Even though most mesenteric cysts are benign, these lesions do occasionally cause complications, including intestinal obstruction, volvulus, or even torsion , 4. In tThe preoperative diagnosis may be achieved with the common imaging examinations of the abdomen . Ultrasonography usually demonstrates a cystic tumour whose content may form a fluid-fluid level in case of a chylolymphatic cyst . A plainDifferent surgical approaches are used, that is, marsupialization, sclerotherapy, drainage, enucleation, percutaneous aspiration, and excision of the cyst with or without resection of the involved gut –12. But  H. capsulatum. H. capsulatum is one of several thermally dimorphic fungi; at 35–37°C in the lungs, it transforms from a mold state into a yeast state [Histoplasmosis is caused by inhaling the microconidia (spores) formed by the environmental mold. Healthy hosts, unless they inhale many conidia, remain asymptomatic or have only mild pulmonary symptoms when exposed tost state .Before immunity develops, in almost all cases, the organism is disseminated by the macrophages moving to local mediastinal and hilar lymph nodes and, more widely, to the liver, spleen, lymph nodes, and other organs. This dissemination is asymptomatic in most individuals but can lead to progressive disseminated infection in people that are immunosuppressed . possibility [ H. capsulatum can be found throughout the GI tract from the mouth to the anus [All gastrointestinal (GI) forms of histoplasmosis are likely manifestations of disseminated disease. Acquisition by the GI route through water or food ingestion has been suggested, but this has never been proven and remains a very unlikelysibility . H. capsthe anus . It is lThe most common presenting symptoms of GI histoplasmosis are abdominal pain and diarrhea , 19. Diaµg/mL. The oral solution is given on an empty stomach. If the patient cannot tolerate the solution, capsules can be substituted. Itraconazole capsules must be given with food and require acid for absorption, so acid-inhibiting drugs cannot be prescribed for these patients [Patients who have mild-to-moderate disease can be treated with oral itraconazole as initial therapy. Itraconazole is best administered as an oral solution at a dosage of 200 mg three times daily for 3 days as a loading dose and then twice daily for 12 months. Itraconazole levels should be monitored after the second week of therapy to be certain that serum levels are at least 2 Most patients with disseminated histoplasmosis respond well to antifungal therapy. This patient is currently on treatment.We cannot confirm whether the chylous cyst was due to the histoplasmosis infection or whether the histoplasmosis secondarily colonized an already existent cyst. But the presentation of these two conditions in the same patient makes it a very rare presentation.


A review of the literature is also presented.Chondrosarcoma, the second most common primary malignancy of the bone, is malignant cartilage forming tumour that very rarely involves the axial skeleton. It may arise as a primary bone tumour or as a secondary lesion from a preexisting benign cartilaginous neoplasm such as osteochondroma or enchondroma. A rare case of a massive secondary lumbar spine chondrosarcoma is presented. Management consisted of an initial posterior spinal stabilization and fusion and then a curative radical  Chondrosarcoma is the second most common primary malignancy of the bone, of which men are more often affected than women –5. ChondA case of a male patient with a large secondary spinal chondrosarcoma arising from the malignant degeneration of a lumbar vertebral osteochondroma is presented and the inherent complex surgical management is discussed, in light of the literature.A 29-year-old male, warehouse worker, presented with a noticeable mass in the left side of his abdomen and weight loss of three and half stones over a four-month period. His past medical history and family history were unremarkable. Physical examination revealed a large, nontender, and fixed left-sided abdominal mass and a normal neurovascular examination except an isolated reduction in light touch sensation in the L2-L3 dermatomes of his left leg only.An abdominal and pelvic magnetic resonance imaging (MRI) and computed tomography (CT) demonstrated a large, 19 × 16 × 11 cm mass arising from the anterolateral aspect of the L3 pedicle with peripheral calcification, completely obliterating the distal psoas muscle and causing a considerable mass effect to surrounding structures, with displacement of the left kidney. Distally, the tumour extended to the level of the pelvic inlet but not into the spinal canal Figures . en bloc tumour resection were utilized.CT guided biopsy of the left abdominal mass confirmed the diagnosis of a low-grade chondrosarcoma arising from an osteochondroma from the lateral body of the L3 vertebrae. The difficult anatomical location and the large size of the lesion greatly increased the complexity and risks of any surgical approach. In order to improve the chances of surgical success and reduce and prepare for possible risks and complication such as catastrophic haemorrhage, spinal instability, and paralysis/neurological deficit, a two-stage surgical procedure and a multidisciplinary team approach to a curative en bloc tumour resection.In the first stage, a posterior midline incision to expose the lumbar spine was performed. Bilateral pedicle screws were placed at the levels of L2 and L4, with rods fixated at the end of the procedure for spinal stabilisation. Bilateral laminectomies and facetectomies were performed leading to exposure of the nerve roots, of which the left L3 nerve root was identified and ligated. A partial osteotomy was carried out at the level of L3 within the vertebral body to allow ease of hemivertebral body resection with the forthcoming en bloc with an intact envelope, surrounding soft tissue and completion of the L3 hemivertebral column resection osteotomy with partial resection of the vertebral bodies of L2 and to a lesser extent L1 and L4. After closure of the anterior wound, the patient was placed prone. The previous posterior midline wound was reopened and extended to expose the thoracolumbar spine from T12 to L4. The original posterior instrumentation was extended to include T12 and L1, with only right unilateral pedicle screws to L2 and L3 and bilateral pedicle screws to T12, L1, and L4. Spinal fusion from T12 to L4 using intervertebral titanium cages filled with allograft and demineralized bone matrix for additional stability was performed. The posterior needle biopsy tract was resected and the large excised tumour was sent for histopathological evaluation, which affirmed the previous biopsy results of a low-grade classical secondary chondrosarcoma with all margins free of tumour , which is an autosomal dominant condition , 18. SpiThe malignant transformation of an osteochondroma to chondrosarcoma is well described, albeit a rare occurrence. The conversion typically results in a low-grade chondrosarcoma, but higher grades are possible. This malignant conversion is normally associated with a sudden increase in the size of the lesion, increased or new-onset pain, an increase in the cartilaginous cap thickness, and recurrence of the lesion after total resection , 17–20. Patients with spinal chondrosarcoma usually present with visible and/or palpable mass, pain, and variable neurological symptoms , 8, 10. ring and arc” calcification pattern [Typically, spinal chondrosarcoma has a malignant appearance radiographically. Plain radiographs usually demonstrate a radiolucent lesion with varying degrees of a cartilage type matrix calcification, usually a “ pattern , 14. CT  pattern . Both MR pattern . In addi pattern , 20. en bloc excision with wide disease free margins. This is usually the treatment of choice, as this provides the potential for cure and a significant reduction in recurrence rates, which is said to be as low as 3 to 8% [Secondary chondrosarcomas are diagnosed histologically but radiological and gross features of the tumour need to be taken into account and correlated. Spinal chondrosarcoma usually belongs to a mesenchymal or clear cell histological subtype. However, our patient displayed a low-grade conventional  histological subtype. For secondary spinal chondrosarcomas there are two surgical options, the first of which involves complete 3 to 8% , 22, 23. 3 to 8% , 20. Che 3 to 8% , 22–24. en bloc excision for spinal chondrosarcomas is fraught with multiple technical difficulties such as the size of the tumour , anatomical constraints , risks of spinal instability, and inflicting new neurological deficits [Completedeficits , 12, 22.deficits , 20. Duedeficits , 23, 25.deficits . en bloc resection with adequate tumour-free margins and at one-year follow-up no sign of recurrence was noted. However, patients with chondrosarcoma may have a delayed course of local recurrence or metastatic disease and long-term follow-up is advised [In our case, we were able to achieve a complete advised , 14.Spinal chondrosarcoma is a rare occurrence and this report depicts a rare and interesting case of the surgical management of a grossly enlarged secondary spinal chondrosarcoma arising from the malignant transformation of a lumbar vertebral osteochondroma with a favourable outcome.


Asymptomatic and oligosymptomatic appendicitis are rare and challenging diagnoses that should not be missed.A young female patient presented with mild to moderate pain in the middle and lower abdomen, and the results of physical examination, including digital rectal examination, were otherwise non-contributory. Ultrasonography demonstrated a marked increase of the outer appendiceal diameter up to 12 mm and a trace of free liquid around the terminal ileum. Subsequent surgical exploration and histological examination allowed for a final diagnosis of putrid, ulcero-phlegmonous, hemorrhagic appendicitis and periappendicitis.Ultrasonography is increasingly used for the diagnosis of appendiceal inflammation, particularly in military medical settings. Increases in the outer appendiceal diameter up to >6 mm under compression have recently been demonstrated to be indicative of acute appendicitis. At a minimum, in cases with doubtful physical examination results, ultrasonography should be considered as an element in the diagnosis of acute appendicitis. The management of appendicitis is a standard situation for military surgeons. Among active component members of the U.S. Armed Forces, the overall incidence of appendicitis was 18.4 per 10,000 person-years between 2002 and 2011 . The higThe use of ultrasound scanning of the abdomen for the diagnosis of appendicitis has previously been evaluated. In cases of acute appendicitis, the outer appendiceal diameter increases to 7.9 ± 2.0 mm compared with a typical outer appendiceal diameter value of 4.5 ± 1.2 mm for healthy populations. Further, periappendiceal inflamed fat is frequently detected by ultrasound scanning in patients with appendicitis [Here, we describe a rare case of progressed appendicitis that nevertheless presented with mild to moderate symptoms and was diagnosed based only on ultrasound scanning results.A 21-year old female patient presented at the emergency department of a hospital with progressive pain in the middle and lower right abdomen, nausea and vomiting. The symptoms began 12 h prior to her arrival. Defecation and stool consistency were non-contributory. Fever and chills were denied. Lactose intolerance was the only reported pre-existing condition.The rectal temperature of the patient was 37.4 °C. The patient was alert and oriented. The described pain in the middle and lower right abdomen could be induced by pressure. Apart from these findings, the physical examination was non-contributory.Specifically, the patient’s abdomen was soft, and there were no signs of guarding or peritoneal signs. Auscultation of the abdomen was non-contributory. A digital rectal examination did not provoke any pain in the recto-uterine pouch. The sphincter tone was normal. The stool was brown and did not exhibit any signs of blood or viscous secretions. The reported pain was noticeably decreased following novaminsulfon infusion. Overall, the patient appeared to be only marginally compromised, and dismissal from the emergency department was therefore considered.9/L (ref.: 3.8 × 109/L – 9.8 × 109/L) was observed, whereas the C-reactive protein level was non-contributory at 2.4 mg/L (ref.: <5). A slightly increased free tetraiodothyronine level of 15.7 pmol/L (ref.: 7.7 – 14.2) was observed as an incidental finding. All other assessed laboratory parameters were normal.A marked increase in the white blood cell count to 17.8 × 10There was a trace of free fluid around the terminal ileum. Further, an intestinal cockade sign was visible, and the size of the appendix wall was increased to 3 mm Fig. . In the The overall accuracy of ultrasonography for the identification of acute appendicitis was recently estimated at approximately 90 % based on an analysis of a small cohort of 60 patients . In thisBased on the ultrasound results, a diagnosis of acute appendicitis was made. Laparoscopic surgical intervention allowed for the extirpation of an inflamed, phlegmonous appendix Fig. .Fig. 2InThe operation and postoperative management were without any complications.Putrid, ulcero-phlegmonous, hemorrhagic appendicitis and peri-appendicitis with putrid fibrinous serositis and fecal impaction were diagnosed. A partial spread of the inflammatory process beyond the surgical margins was observed.Skilled use of abdominal ultrasonography can serve critical diagnostic purposes for forward deployed troops when other modalities, such as computed tomography, are not available due to the austere settings. In such situations, abdominal ultrasonography is particularly important for young females for whom the differential diagnoses of abdominal and pelvic pain are much broader , 8 and iThe peculiarity of the present case is that ultrasonography provided the most important information for the diagnosis of appendicitis. Oligosymptomatic and asymptomatic cases of appendicitis are both rare  and chalBoth computed tomography (CT) and ultrasound scanning (US) have been demonstrated to be useful modalities in the diagnosis of acute appendicitis. Both techniques are used to reduce the number of unnecessary surgical interventions , but US CT is the most accurate imaging modality for cases of suspected appendicitis and should be considered in cases of questionable US findings when a CT scanner is available . If ther2-weighted images has been demonstrated to be an expensive but useful alternative for the detection of acute appendicitis [If ionizing radiation is to be avoided, magnetic resonance imaging involving gadolinium-enhanced and Tndicitis , 31. Recndicitis . Larger In cases of less advanced oligosymptomatic appendicitis, colonscopy is an alternative procedure that might support the diagnosis . In contAs demonstrated by a case of bilateral ureteral obstruction following asymptomatic appendicitis , the conWritten informed consent for the publication of this case report and any accompanying images was obtained from the patient. A copy of the written consent is available for review by the Editor-in-Chief of this journal.CT, computed tomography; US, ultrasound scanning


Endoscopic ultrasound fine-needle aspiration (EUS-FNA) is used to make a cytopathologic diagnosis of suspicious lesions located around the gastrointestinal tract. It is a safe technique with few complications. The most common complications of EUS-FNA are related to pancreatic lesions . Rare complications have been noted such as stent malfunction, air embolism, infection, neural and vascular injuries, and tumor cell seeding. There are very few studies examining equipment malfunctions. We report a case of needle fracture during the EUS-FNA of suspicious thoracic lymph nodes in a 79-year-old man investigated for unexplained weight loss. Endoscopic ultrasound fine-needle aspiration (EUS-FNA) is used to make a cytopathologic diagnosis of suspicious lesions located around the gastrointestinal tract. These include mediastinal lymph nodes easily accessible with the transesophageal position. It is a safe technique with few complications. A recent review article  found anA 79-year-old man, past smoker, known for hypertension and diabetes, was hospitalized for an unexplained weight loss. During the initial investigation, multiple lymphadenopathies were discovered on a thoracic scanner including lymph nodes located anterior to the trachea (2.1 × 2.5 cm), in the aortopulmonary window (1.5 × 3.5 cm), inferior to the carina (1.5 × 3.5 cm), and at the right and left hilum measuring, respectively, 3.5 × 2.7 cm and 3 × 1.2 cm. In addition, peribronchial micronodules were located in the right and left superior lobes, the inferior right lobe, and the root of the middle lobe. A bronchoscopy was performed. Reactive cellular changes without malign neoplastic cells were found on the bronchial washing, while the bronchial biopsy demonstrated nonnecrotizing granuloma inflammation. Multiple possible diagnoses were retained including lymphoma, non-small cell pulmonary neoplasm, sarcoidosis, and tuberculosis.™ Slimline Needle, Boston Scientific) [™ Slimline Needle, Boston Scientific) [Under conscious sedation, an EUS was performed to biopsy a 30 × 15 mm, partially calcified, hypoechogenic subcarinal lymph node  . During entific) . An inadentific) . Signifientific) . After sentific) , and an entific) . The tipentific) . The neeentific) . Two endentific) .A thoracic scan following the procedure showed a mild paraesophageal pneumomediastinum and a discreet 29 × 4 mm parietal hematoma, but no signs of perforation with ingestion of contrast. The patient was completely asymptomatic and did not require any surgical intervention. A control CT scan showed no oral contrast extravasation and complete resolution of the pneumomediastinum within 24 hours. The patient was then able to eat without sequelae. The biopsy specimen demonstrated a few small nonnecrotizing granulomas, epithelioid histiocytes, and rare giant cells. An infectious process or sarcoidosis was retained as possible diagnosis .Many authors have explored the complications associated with EUS-FNA –3, but oIn their study, comparing the effectiveness of the 22-gauge and 25-gauge needles in the diagnosis of pancreatic solid mass, Siddiqui et al.  mentioneThere is more literature on needle fractures in pulmonary medicine. In fact, a recent survey examining complications associated with endobronchial ultrasound-guided transbronchial needle aspiration (EBUS-TBNA) done by the Japan Society for Respiratory Endoscopy  found nePresently, the consequences of a broken needle during ultrasound-assisted techniques are unknown due to the limited literature on the subject. It is possible to assume that if the foreign body is left in situ, an inflammatory reaction may develop within the tissue leading to further complications. In addition, there is always the possibility of the fragments dislodging and migrating while damaging the mucosal lining. This may result in perforation and surgical intervention. It is difficult to identify specific risk factors for needle fracture. However, as mentioned by other authors, certain endoscopic maneuvers as torqueing, multiple punctures with the same needle, and rigid tissue texture could all be predisposing factors. We also feel that repeated erector movement for the fanning technique in the hard lymph node may have caused a focal weakness on the needle in our case. It is important for clinicians to be aware not only of complications associated with ultrasound-guided techniques, but also of complications related to the devices themselves including fractured needles.


A focused bedside ultrasound over the patient’s region of maximal discomfort revealed a thickened bowel wall and several small contiguous hypoechoic projections surrounding a hyperechoic center, suggestive of diverticulitis (A 72-year-old otherwise healthy female presented to the emergency department with two weeks of worsening abdominal pain. She was afebrile with normal vital signs. Her physical examination was notable for moderate abdominal tenderness without rebound to the left and suprapubic regions of the abdomen. Laboratory studies were remarkable for a white blood cell count of 13,000/mmiculitis . She wasiculitis .Acute diverticulitis resulting from inflammation of colonic diverticulum affects over half the population greater 65 years of age.5Particularly in cases of suspected uncomplicated diverticulitis, abdominal ultrasound may reach the diagnostic reliability of CT.2


Melorheostosis is a rare entity belonging to the group of sclerotic bone dysplasias. Described for the first time in 1922 by Leri, it remains imperfectly known as clinical presentations are highly variable, and the etiological diagnosis is not fully elucidated. We report a case of polyostoticmelorheostosis for which radiological investigations were complete, in order to study this disease. Melorheostosis is a rare benign non hereditary condition. It is known as a mesoderm sclerotic bone dysplasia, characterized by cortical hyperostosis with or without retraction of soft tissue. It was described for the first time in 1922 by Leri and Joanny as a dripping candle wax hyperostosis –5. The dWe report the case of a 39 years-old female, with no past medical history, whose complaint was a spontaneous one week evolving isolated left cruralgia. The clinical examination was unremarkable apart from an anterior thigh pain at the mobilization of the left hip. No quadriceps hypotrophy, no fever and no limitations in the joints motion were found. Plain radiographs of the pelvis and proximal femur showed hypercondensation of the medial cortex of the left femur below the calcar, the classic flowing candle wax, combined with condensation of the iliac side of the sacroiliac joints bilaterally . Lab tesMelorheostosis is still an imperfectly known pathology and many questions remain unanswered. The role of a genetic predisposition has been suggested, but evidence does not yet exist considering that familial cases have not yet been reported. Its association with vascular sector malformations and some observations reported changes in skin pigmentation suggests that a mutation may be responsible –5.In this rare disease, the clinical presentations are highly variable and locations are diverse. Both upper or lower limbs can be affected in addition to the axial skeleton. The lower limbs are affected more often than the upper limbs. Conventional imaging is very characteristic when it shows the flowing candle wax, but this aspect can be seen in other conditions such as osteomyelitis or bone tumor. In some presentations it may be lawful to biopsy and perform bacteriological examinations . In the The therapeutic is not well codified. It is symptomatic in most cases, in order to control bone pain. Protocols based on Pamidronate  have beeMelorheostosis is a rare, benign with chronic course, often with outbreaks interspersed with remissions. We emphasize the need to ensure the diagnosis by plane radiographies, CT scan or MRI. If any doubt, biopsy may be necessary. Scintigraphy is the best exploration for monitoring the disease. Treatment is symptomatic essentially, based on nonsteroidal anti-inflammatory. Surgery can have its place in large deformations and joint damage, but with great care.