Odontogenic keratocyst (OKC) is a common developmental odontogenic cyst affecting the maxillofacial region. Multiple OKCs are usually seen in association with nevoid basal cell carcinoma syndrome (NBCCS) but approximately 5% of patients with OKC have multiple cysts without concomitant syndromic presentation. This report represents a case of multiple OKCs in a non-syndromic patient Odontogenic keratocyst (OKC) is a developmental odontogenic cyst with specific histopathologic features and clinical behavior. Several investigators suggested that OKCs must be regarded as a benign cystic neoplasm rather than a cyst.In the latest WHO classification of odontogenic tumors in 2005, these lesions have been given the name “karatocystic odontogenic tumors” (KCOTs) . Multiple KCOTs are usually seen with cutaneous, skeletal, ocular and neurologic abnormalities as a component of nevoid basal cell carcinoma syndrome (NBCCS). The features of this syndrome were first described by Gorlin and Goltz in 1960, so it is also recognized as Gorlin- Goltz syndrome .KCOTs in the jaws arise from the cell rests of dental lamina and usually are seen during the second to fourth decades of life with a slight male predilection . TypicalThis study reports a case of multiple KCOTs without any syndromic manifestations.An 11- year-old boy with a complaint of swelling in left side of the upper jaw was referred to our clinic. Systemic signs and symptoms, past medical history and hematologic tests were within normal limits. The radiographies from chest and skull were unremarkable and no cutaneous abnormality was revealed. In panoramic radiograph, two radiolucencies with corticated border were revealed around the unerupted mandibular left canine and the unerupted maxillary left second molar area. Maxillary second molar was displaced .Regarding the radiographic examination and presence of unerupted teeth and their location, the initial differential diagnosis was dentigerous cyst and the second was KCOT. Other odontogenic cysts and tumors such as adenmatoid odontogenic tumor were considered as other differential diagnoses.Enucleation of the cystic lesions was performed under local anesthesia and tissue samples were obtained for histopathologic examination. The surgical specimens were sheet- like with cystic appearance. After processing, the tissue samples were sectioned and stained with hematoxylin and eosin (H&E). The histopathologic examination revealed that the cystic lining of mandibular lesion was corrugated parakeratinized epithelium with uniform thickness of 5-6 peg formation. The cyst wall was composed of a non-inflammatory fibrous connective tissue. All these features established the diagnosis of KCOT for the mandibular lesion . But theWith definitive diagnosis of KCOT in mandibular lesion and absence of any evidence of NBCCS in clinical examinations, multiple sections were cut from the maxillary lesion. These sections were obtained because of the probability of multiple non- syndromic KCOTs.H & E slides were evaluated carefully and at last a small epithelial lining with characteristic features of KCOT was detected .According to these entire features which were correlated with the clinical and radiographic findings, the diagnosis of KCOT was established for both cystic lesions.OKC is a common developmental odontogenic cyst and its biologic behavior is similar to a benign neoplasm . TherefoKCOT may be found in any age with peak prevalence between 10 to 40 years old .The mandible is involved in 60 to 80% of cases with a marked tendency to occur in the posterior body and ascending ramus , 7.Small KCOTs are usually asymptomatic but larger ones may show clinical manifestations like pain, swelling or drainage . The preRadiologically, KCOTs demonstrate a well defined radiolucent area with smooth and often corticated margins and may be unilocular or multilocular. In 25 to 40% of cases, an unerupted tooth is seen in association with the lesion . RadiogrIn this case, microscopic evaluation of the mandibular lesion showed characteristic features of KCOT inducing a corrugated parakeratinized stratified squamous epithelial lining with palisaded basal cell layer without rete ridge formation .Maxillary lesion showed histopathologic features of an inflammatory odontogenic cyst, but regarding the possibility of multiple KCOTs in this patient, several sections were prepared to achieve the correct diagnosis.Finally we revealed characteristic features of KCOT in small area of histopathologic slides and the diagnosis of other cysts, especially inflammatory dentigerous cyst, was ruled out.Finally, the diagnosis of KCOT was established for both cystic lesions.Multiple KCOTs are usually considered as a component of Gorlin- Goltz syndrome or NBCCS , orofaciRarely, multiple KCOTs are seen without other syndromic manifestations .Brannon  reportedNBCCS is recognized by multiple KCOTs, nevoid basal cell carcinomas of the skin, bifid ribs, calcification of the falx cerebri and other features .However, except for presence of KCOTs, our patient was healthy in clinical examinations and suggestive features of these syndromes such as basal cell carcinoma, skeletal or orofacial defects, stunted growth, bleeding diathesis, hyper-extensible skin and hypermobile joints and other features were not present. Multiple KCOTs might be the first and the only manifestation of NBCCS without any other features associated with syndrome. However, other symptoms can occur in later decades of life .Similar cases to the current case have been reported in a few published English articles.Auluck et al.  discusseSholapurkur et al.  presenteParikh  reportedBartake et al.  reportedGuruprasad et al.  discusseAlso, findings of Habibi et al.  study onTherapeutic interventions of KCOT include marsupialization and enucleation, combined with adjuvant cryotherapy with Carnoy’s solution, and marginal or radical resection .For unerupted permanent teeth in children, conservative treatment should be done first, because an aggressive operation can cause adverse effects on teeth development and its eruption. Marsupialization followed by enucleation has the lowest recurrence rate among the conservative treatments .KCOTs related to NBCCS have more aggressive behavior and higher recurrence rates than non- syndromic ones.This characteristic is due to a high proliferation rate of the epithelial linings in the syndromic cases .There is a little information about the relationship between cell proliferative markers and the recurrence rate of KCOTs. The study performed by kuroyanagi et al.  showed tSince our patient lived in a small village, following up and checking the recurrence of the lesions was impossible.In any patient with a KCOT, the presence of multiple KCOTs should be considered. Therefore, careful histopathologic examination for any other existing lesion should be done. Moreover, a complete clinical examination and long- term follow up must be performed to detect any other features associated with NBCCS.


We report the first known case of a retroperitoneal leiomyosarcoma that presented with an endoscopically defined source of gastrointestinal bleeding in the colon. A 68-year-old male with a history of diverticulosis, hypertension, and hypercholesterolemia who complained of a 3-month history of abdominal pain, nausea, and intermittent hematochezia presented for evaluation of large volume hematochezia and lightheadedness. Colonoscopy revealed left-sided diverticulosis and rectal varices without stigmata of recent bleed. CT scan showed a 26 × 20 × 13 cm heterogeneous retroperitoneal mass and multiple hypodense hepatic lesions. Liver biopsy revealed leiomyosarcoma. In summary, although surgery is the mainstay of treatment, resectability has not improved significantly. Early recognition and aggressive surgery are keys to long-term survival. Retroperitoneal soft-tissue sarcomas rare. They are locally invasive, large tumors that usually present with abdominal discomfort and palpable mass. They rarely present with lower gastrointestinal bleeding. We report the first known case of a retroperitoneal leiomyosarcoma that presented with an endoscopically defined source of gastrointestinal bleeding in the colon.We present a case of a 68-year-old male with a history of diverticulosis, hypertension, and hypercholesterolemia who complained of a 3-month history of abdominal pain, nausea, and intermittent hematochezia. He presented to the emergency room for evaluation of large volume hematochezia and lightheadedness. Physical examination revealed a systolic blood pressure of 80 mmHg. Abdominal exam showed mild distension and diffuse tenderness. Nasogastric lavage was clear. Rectal exam was notable for bright red blood, but no mass or hemorrhoids. Laboratory evaluation showed blood urea nitrogen (BUN) = 35 mg/dL, creatinine = 1.9 mg/dL, international normalized ratio (INR) = 1.4, hemoglobin = 4.8 g/dL, and platelet count = 498,000 k/mcl. Upper endoscopy revealed a hiatal hernia and extrinsic compression of the second part of the duodenum, but there were no signs of active or recent bleeding. Colonoscopy revealed left-sided diverticulosis and rectal varices without stigmata of recent bleed. Additionally, a large cavity was found in the proximal ascending colon with a necrotic mass and surrounding varices . No actiThe patient underwent exploratory laparotomy for palliative right hemicolectomy and was found to have a large tumor extending from the right retroperitoneum lateral to the right colon and anterior to the right kidney. The mass was invading the colonic mesentery and encased the proximal one-third of the superior mesenteric artery. Therefore, he was deemed inoperable and was discharged home with hospice care.Soft-tissue sarcomas are rare accounting for approximately 1% of adult malignancies. Fifty percent of these occur in the extremities followed by retroperitoneal/visceral and trunk involvement . Most caThere have been 36 cases reported of duodenocaval fistula and one case of gastric invasion of retroperitoneal sarcomas causing gastrointestinal bleeding , 5, all The overall 5-year survival rate of retroperitoneal sarcomas 36%–58% and is dependent on tumor histology and extent of tumor invasion . SurgeryIn contrast to the promising results seen with extremity soft-tissue sarcoma and childhood sarcomas, the addition of adjuvant radiotherapy or chemotherapy has not altered prognosis. In a review of 312 patients with abdominal and truncal sarcomas treated from 1977 to 2004, the ability to resect the lesion, negative resection margins, and tumor histology were the only good prognostic factors . LiposarEarly recognition and aggressive surgery are the keys to long-term survival of patients with retroperitoneal sarcomas, but the mainstay of treating advanced disease is yet to be determined and needs further research as the overall prognosis of advanced disease is relatively poor.


Granulosa cell tumor (GST) is a sex-cord/stromal neoplasm of the gonads, more commonly arising in the ovaries, while approximately 80 cases have been reported in the testes. Out of these, 30 cases were of the adult type, while the remainder 50 cases were of the juvenile type. The latter mostly concerned infants and followed a benign course. However, the adult type testicular GCTs may be potentially malignant as it also happens in female patients with such neoplasms. We present a case of an adult type GCT located at the left testis. The patient was subjected to total orchiectomy and received no further treatment. Histology showed typical GCT histomorphology with Call-Exner bodies in some places. The immunoprofile of the tumor was CD99 (+), calretinin (+), inhibin (+), alpha smooth muscle actin (+), vimentin (+), ER (−), PR (−), keratin AE1/AE3 (−), alpha fetoprotein (−), CD117 (−), and placental alkaline phosphatase (−). Two years after surgery, the patient is alive and well with no signs of recurrence. Granulosa cell tumor (GST) belongs to the sex-cord/stromal tumors of the gonads. Two forms of GST have been recognized, namely, the typical adult type, and its variation, and the juvenile type. GST arises far more commonly in the ovaries but on rare occasions may be encountered in the testes. Most of the latter cases concerned infants, which presented the juvenile type morphology –3, while A 37-year-old white male attended our hospital for scrotal swelling. He did not present any signs and symptoms suggestive of a hormone-secreting tumor like gynecomastia, and his history was otherwise unremarkable. Palpation, ultrasound examination, and MRI revealed a tumor of the left testis , and so The surgical specimen weighted 106 gr and consisted of the testis measuring 6.6 × 6 × 4 cm, the epididymis 2.5 in length, and the spermatic cord measuring 9.2 × 2.5 × 2.5 cm. On cross-sections, the testis presented a whitish, solid tumor measuring 4.2 × 2 × 3.1 cm. Histology showed a sex-cord tumor of the testis . The tumGCTs include tumors composed of granulosa cells, theca cells, and fibroblasts in varying degrees and combinations. GCTs account for approximately 2% of all ovarian tumors and can be divided into the much more frequent adult type (95%) and the juvenile type (5%), based on histologic findings. The opposite seems to occur in males where the majority of the reported cases (50/80 cases) belong to the juvenile type of GST, while only 30 cases in the literature correspond to the adult type GST –10. The Adult type GSTs also manifest with scrotal swelling, even though a minority of cases has presented gynecomastia –10. The Very little is known about the histogenesis and etiology of GCTs, especially in the testes. Recently the FOXL2 gene has been implicated in the pathogenesis of the adult type GST of the ovaries. FOXL2 is a transcription factor, which plays important role in the development of normal ovaries and is a key factor in female sex determination . A misseOvarian adult GCTs are considered as potentially malignant neoplasms. Approximately 20% of these tumors will eventually recur or metastasize, even many years after the initial diagnosis . In a siIn conclusion, our report highlights one more case of this very rare tumor of the testis, which is quite problematic in terms of prognosis and management, and for this reason seems to have attracted the interest of many researchers recently. Long-term followup is recommended, since recurrence of the disease may appear late in the clinical course.


Sir,Wuchereria bancrofti accounts for 90% of the total filarial infections.[Filariasis is a major health problem of the tropics and subtropics, commonly seen in countries like India, China, Indonesia, Africa, and the Far East. The disease is endemic all over India. In Indiafections. Despite fections.–6 MicrofW. bancrofti having multiple, coarse, discrete nuclei extending from the head to the tail, except in the small terminal portion of the caudal end [W. bancrofti was offered. The peripheral blood smear did not reveal any microfilariae. The patient was advised chest radiographs, which revealed a mass lesion in the upper lobe of the left lung.A 45-year-old male presented in the outpatient department with supraclavicular and cervical lymphadenopathy since 3 months. The left supraclavicular lymph node was of size 3cm×3cm and the left cervical lymph node was of size 2cm×2cm. The patient was referred to the pathology department for FNAC of the left supraclavicular lymph node. Microscopic examination showed highly cellular smears. Epithelial cells were seen arranged in groups, acinar pattern, and scattered singly. Cells were fairly uniform in size with round, hyperchromatic nuclei [It is estimated that about 600 million people are living in areas endemic for lymphatic filariasis in the Southeast Asian region. There are about 60 million people infected in the region and about 31 million people have clinical manifestation of the disease.[Filariasis is a major public health problem in India. Heavily infected areas are found in Uttar Pradesh, Bihar, Jharkhand, Andhra Pradesh, Orissa, Tamilnadu, Kerala, and Gujarat. The disease manifestation ranges from none to both acute and chronic manifestations.W. bancrofti and Brugia malayi, which are responsible for 90% and 10% of the cases, respectively.[μ long), which dwell in the peripheral blood for 5–10 years.[It is transmitted by the Culex mosquito and is caused by two closely related nematodes, ectively. Infectiv10 years.Despite the high incidence of filariasis, microfilaria in FNAC is not a very common finding. There have been reports of single or small number of cases of microfilaremia at various sites such as lymph node, breast lump, bone marrow, bronchial aspirate, nipple secretions, pleural and pericardial fluid, ovarian cyst fluid, and cervicovaginal smears.Microfilariae and adult filarial worms have occasionally been detected in association with neoplastic lesions in cytological smears. On searching the literature for microfilaria with malignant neoplasm, we have found some case reports describing coexistence of microfilaria with primary malignant tumor.–5We have not found any case of microfilaria with secondary deposits. The presence of microfilariae along with neoplasms is generally regarded as a chance association.In the present case, we feel that the presence of microfilaria in association with the metastatic deposits is an incidental finding and that the patient was harboring subclinical filariasis when the tumor metastasized. No microfilariae were detected in the peripheral smears. Our case illustrates the coexistence of microfilariae with secondary deposits of adenocarcinoma, which is a rare finding. This also highlights the importance of screening smears for parasites even in the absence of clinical symptoms, particularly in highly endemic areas.


Congenital posterior urethral diverticulum is an uncommon anomaly, sometimes complicated by infection or calculi formation. A conventional voiding cystourethrogram (VCUG) is the most commonly used diagnostic modality. Dynamic magnetic resonance imaging (MRI) has not been frequently described in this entity. We describe a case of posterior urethral diverticulum complicated with secondary calculi, where the patient was evaluated using dynamic MRI and conventional VCUG. Congenital urethral diverticula in boys are extremely rare, especially those arising from the posterior urethra.–3 We repEscherichia coli. USG revealed a normal right kidney, an empty left renal fossa, and two cystic swellings in the pelvis that were communicating with each other through a narrow channel The posteriorly located cystic swelling contained echogenic contents, with acoustic shadowing suggestive of calculi. The left kidney could not be visualized. We suspected a urinary bladder diverticulum or a midline cystic lesion such as a Mullerian duct/prostatic utricular cyst. The absent left kidney was confirmed with a renal Tc99m dimercaptosuccinic acid (DMSA) nuclear scan [An 11-year-old male child presented with complaints of post-void dribbling of urine and pain in the abdomen for the last two years. Physical examination revealed bilateral empty scrotal sacs, with a normally developed phallus. Laboratory evaluation revealed a normal hemogram and normal renal function tests. Microscopic examination of urine showed pus cells, and culture grew ear scan .Subsequently, a voiding cystourethrogram (VCUG) was performed. The plain radiograph revealed multiple calculi in the pelvis, overlying the symphysis pubis . The VCUMRI was then performed on a 1.5-T system  for delineation of the anatomy and the associated anomalies. Routine T1W  and T2W images with fat suppression  were acquired using a 4-mm slice thickness and a 256 × 256 matrix. The T2W axial image of the pelvis revealed a fluid-filled structure posterior to the bladder . The rigCystourethroscopy revealed the opening (neck of the diverticulum) in the prostatic urethra. A surgical resection through a perineal approach was performed, and histopathology confirmed the presence of transitional epithelium in the diverticulum.A urethral diverticulum is a sac-like outpouching of the urethral mucosa or simply a tubular sac connecting into the urethra at any point in its course. It is more common in females than males.–3 These Unlike anterior urethral diverticula, the majority of posterior urethral diverticula are acquired in origin. PatientsUSG and VCUG are usually the initial modalities of choice and are adequate in most cases. However, a giant posterior urethral diverticulum may cause diagnostic difficulties due to distorted anatomy, and additional imaging may be required for proper preoperative assessment.MRI, because of its good soft tissue contrast resolution, allows superior visualization of the urethra and the adjacent structures. It is also possible to visualize the diverticulum sac which may not be opacified on contrast studies. Dynamic 7In conclusion, the excellent soft tissue contrast, the lack of exposure to radiation, and the good temporal resolution that the newer generation fast gradient-echo sequences provide make dynamic imaging possible. MRI can be the ideal imaging modality for evaluation of the genitourinary tract in children.8 Dynamic


Primary angiosarcoma of the brain is extremely rare; only 15 cases have been reported in adults over the last 25 years.We describe two cases of primary angiosarcoma of the brain that are well characterized by imaging, histopathology, and immunohistochemistry. Case 1: our first patient was a 35-year-old woman who developed exophthalmos. Subtotal resection of a left extra-axial retro-orbital mass was performed. Case 2: our second patient was a 47-year-old man who presented to our facility with acute visual loss, word-finding difficulty and subtle memory loss. A heterogeneously-enhancing left sphenoid wing mass was removed. We also review the literature aiming at developing a rational approach to diagnosis and treatment, given the rarity of this entity.Gross total resection is the standard of care for primary angiosarcoma of the brain. Adjuvant radiation and chemotherapy are playing increasingly recognized roles in the therapy of these rare tumors. Mena l.[et al. also repl.[et al.-9  inhibitor, has been used with some success in recurrent glioblastoma, a primary CNS tumor that is known to produce VEGF . The useet al.. Median In addition, two multicenter phase II clinical trials are currently in recruitment to look at paclitaxel and bevacizumab in the treatment of metastatic or unresectable visceral or soft tissue angiosarcoma (NCT01055028 and NCT01303497). These studies do not address primary CNS angiosarcoma; in fact, known CNS disease is an exclusion criterion.The rarity of primary angiosarcoma of the brain and the resulting paucity of relevant studies and case reports precludes a definitive judgment concerning optimal therapy. Regardless, suggestions derived from the few published cases, along with the short follow-up from our two more recent cases, may allow the preliminary formulation of a potentially effective treatment plan using modern, multimodality therapy. Surgery with or without tumor embolization, with the goal of gross total resection, remains the standard of care. Post-operative adjuvant radiation therapy may provide a benefit, particularly in cases where gross total resection is not possible. There may be a rationale for using anti-angiogenesis agents such as bevacizumab, perhaps in combination with temozolomide for synergistic anti-angiogenic effect. Finally, treatment results for paclitaxel in soft tissue angiosarcoma suggest that there may also be a role for this chemotherapeutic agent in primary CNS angiosarcoma.Written informed consent was obtained from both patients for publication of this manuscript and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.JRH drafted the manuscript; CAP provided the neuropathology and participated in the final draft; KOR was responsible for the neurosurgical care of both our patients; JKC provided the neuroradiology interpretations for both our patients as well as this section in the manuscript; HMFS was responsible for one of our patient’s chemotherapeutic regimens and helped to draft the manuscript; LBN conceived the study, was responsible for one of our patient’s chemotherapeutic regimens and helped to draft the manuscript. All authors read and approved the final manuscript.


Choriocarcinoma is a very rare cause of ocular metastasis. Only 18 male patients have been reported on, 4 of whom survived, but with significant loss of vision.A 26-year-old Caucasian man, suffering from testicular choriocarcinoma with pulmonary, cerebral, renal, hepatic and osseous metastases, underwent left radical orchiectomy. While being treated with chemotherapy, he presented with loss of vision in the left eye. Ophthalmoscopy revealed bilateral non-pigmented, hemorrhagic choroidal tumours, compatible with secondary lesions. Continued chemotherapy and stereotactic radiotherapy of the skull and spine lead to full remission with excellent vision, after more than 4 years of follow up.Testicular choriocarcinoma is an exceptional cause of choroidal metastasis, potentially asymptomatic and with specific clinical features. Radiotherapy can complement radical orchiectomy and chemotherapy, to achieve full remission and maintain good vision. Intraocular metastases are present in up to 10% of patients with systemic malignancies . In femaChoriocarcinoma is a highly malignant metastatic tumour, derived from undifferentiated placental trophoblasts  or from non-placental totipotent cells, present in the testis, the ovary or the pineal gland . It is characterized by the large amounts of chorionic gonadotropin produced.Choriocarcinoma is a rare cause of choroidal metastasis. Only 18 male patients have been reported on, and most of them had a poor prognosis -4. We prA 26-year-old man was referred to the Jules-Gonin Eye Hospital with loss of vision and a non-pigmented tumour in his left eye. In the previous two weeks, he had been diagnosed with a left testicular tumour and multiple metastases to the lung, brain, cerebellum, kidney, liver and spine. Histopathology following left radical orchiectomy had revealed a non-seminomatous testicular choriocarcinoma, positive for β-human chorionic gonadotropin (β-HCG) and pancytokeratin, slightly positive for α-fetoprotein (AFP) while negative for carcino-embryonic antigen (CEA). Serum levels were for β-HCG 271 052 U/l ), for AFP 0.9 ng/ml  and for LDH 1757 U/l . He was receiving chemotherapy with bleomycin, etoposide and cisplatin.Visual acuity was 6/6 in the right eye and counting fingers (CF) in the left eye. The left posterior fundus showed a large amelanotic choroidal tumour associated with a hemorrhagic retinal detachment. On B-scan ultrasonography, thickness was 7 mm. In the right inferior choroid, a second non pigmented, flat lesion was also present, compatible with a diagnosis of bilateral choroidal metastases Figure .Chemotherapy was continued, with a total of 4 cycles over 3 months' time. Additionally, a stereotactic radiotherapy of the spine and head was performed, the latter including the posterior part of both eyes, using two lateral accelerated photon fields of 6 MV with a total dose of 30.80 Gy in 14 fractions.After four years' follow-up, the patient is in remission without any evidence of metastases. All tumour markers are within normal ranges . Visual acuity is 6/6 in the right and 6/9 in the left eye, both fundi showing a flat chorioretinal scar (Figure We describe a rare case of a patient with bilateral choroidal metastases secondary to testicular choriocarcinoma, successfully treated with chemo- and radiotherapy.MacDonald described the first case in 1936 . A meta-Unlike other metastatic ocular disease, significant haemorrhage is associated with choriocarcinoma metastases in more than 85% of cases and should raise suspicion for this primary -4. KavanPrognosis of testicular germ cell tumours -including choriocarcinoma- was poor until the introduction of cisplatin-based chemotherapy in the mid-1970s . Of the Visual acuity of the 4 surviving patients, who all had unilateral choroidal metastases, ranges between CF and 6/12 ,7-9. OurWe describe a rare case of a patient with testicular choriocarcinoma presenting bilateral choroidal metastases, with specific clinical features and one eye being asymptomatic. Stereotactic radiotherapy of the skull and spine complemented radical orchiectomy and chemotherapy. After more than 4 years of follow up, he is in full remission and has excellent vision.Written informed consent was obtained from the patient 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.The authors declare that they have no competing interests.IG and AS drafted the manuscript. IG, LZ and AS cared for the patient. All authors reviewed the report and approved the final version of the manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2490/11/24/prepub


Paratesticular leiomyosarcomas are rare and can lead to significant morbidity and mortality, if inadequately diagnosed or treated. We present a case of paratesticular leiomyosarcoma in an 88-year-old man presenting with a left scrotal mass with ultrasound revealing that the mass is extratesticular. Left radical orchidectomy was performed and pathological examination of the resected specimen confirmed the diagnoses of high grade leiomyosarcoma with surgical margins clear of tumour. The patient was free of metastatic disease on further imaging and has been disease-free for 18 months. A review of the literature regarding paratesticular leiomyosarcoma presentation, diagnosis, and treatment is also discussed. We report the first documented case of paratesticular leiomyosarcoma diagnosed and treated in Australia. A review of the literature regarding paratesticular leiomyosarcoma presentation, diagnosis, and treatment is also discussed.An 88-year-old male presented to his urologist with a tender swelling of his left testicle. There were no associated lower urinary tract or constitutional symptoms. His past history included prostate adenocarcinoma diagnosed 13 years prior to presentation, for which he had undergone transurethral resection of the prostate and regular monitoring. Physical examination revealed a 2 cm swelling arising from the upper pole of the left testicle. The mass was subcutaneous, firm, and tender. Scrotal ultrasonography identified a left sided hydrocele as well as an altered area of echogenicity at the upper pole of the left testis extending into the epididymis measuring approximately 15 mm in diameter. Prostate specific antigen was unchanged from previous measurements.The patient underwent an elective left radical orchidectomy. The surgical specimen weighing 75 g consisted of testis, epididymis, and spermatic cord. It contained a 35 × 30 × 26 mm firm mass centred on paratesticular tissues adjacent to the upper pole of the left testis extending into the epididymis with a grey/white cut surface. Histologically, the tumour consisted of interweaving fascicles of cytologically malignant spindle shaped cells  with freSoft tissue sarcomas of the genitourinary tract are rare. The American Cancer Society estimates that there were 11,280 new cases of soft tissue sarcoma diagnosed in the United States in 2012, accounting for less than 1% of all new cancer cases . The parPeak incidence is in the sixth and seventh decade . Typical en bloc excisions of all potentially contaminated surrounding soft tissues aiming to obtain negative margin status as well as performing wide inguinal re-resection of soft tissue and scar excision in patients found to have inadequately resected disease [Due to the limited number of cases of this rare malignancy, an ideal treatment protocol has yet to be established with most documented treatments for paratesticular leiomyosarcoma grouped with those for other paratesticular sarcomas. The standard primary treatment is radical orchidectomy with high ligation of the spermatic cord and wide local resection of all nonvital structures. However, due to anatomical constraints, wide circumferential resection margins are rarely achieved and locoregional recurrence after definite surgery is common, occurring with a frequency of approximately 30–50% , 18–21.  disease . Current disease , there iP < 0.01) [There is some evidence supporting the use of adjuvant radiotherapy for paratesticular sarcomas , 23, 24. < 0.01) . It shouThere is currently no clear role for adjuvant chemotherapy in the treatment of paratesticular leiomyosarcoma. A meta-analysis of 14 randomised trials of sarcomas at various sites showed that doxorubicin-based adjuvant chemotherapy led to an improvement in time to local and distant failure . A trendLeiomyosarcoma should be considered as a differential diagnosis in any elderly male presenting with an intrascrotal mass. Although primary management has previously been based on radical orchidectomy with high ligation of the spermatic cord, locoregional recurrence rates are as high as 50%. Adjuvant radiation therapy can lead to improved locoregional control and has a role in patients with nonmetastatic paratesticular leiomyosarcoma. The limited number of cases of this rare disease as well as the inconsistent management strategies utilised requires that further research be performed to formulate an ideal treatment protocol.


Metanephric adenomas are a rare neoplasm of the kidney with less than 200 cases reported. We report a case of a metanephric adenoma incidentally found on imaging in a 52-year-old Hispanic female and treated with robot-assisted laparoscopic partial nephrectomy. A brief review of the literature is also included. Metanephric adenomas of the kidney are a rare and most often benign neoplasm. They are most often removed surgically. The clinical presentation is similar to malignant renal masses and includes polycythemia, hematuria, abdominal pain, and a mass found incidentally on imaging. Less than 200 total cases have been reported to date, and though benign, an increase in the familiarity of this pathology may lead to less invasive treatments in the future.The patient is a 52-year-old Hispanic female that was referred to our clinic for an incidental left anterior midpole hypodense renal mass found on CT following a motor vehicle collision. The R.E.N.A.L nephrometry score of the mass was a 5a, with a size of 19 mm in the greatest dimension . The CT The patient was consented for a left robot-assisted laparoscopic partial nephrectomy. She underwent surgery without any acute complication or difficulty. The estimated blood loss was less than 100 mL, and the clamp time was thirty-eight minutes. She did well over her three-day hospital stay with no complications. Final pathology showed a benign metanephric adenoma (MA). Gross pathology demonstrated a 2.8 by 1.8 by 1.7 cm well-circumscribed, soft, and white-gray mass with a cut surface that was focally friable. The margins were negative. The mass did not penetrate through the renal capsule. The micrographs of our patient's tissue can be seen in the following figures, and were the primary source of our diagnosis. Findings included hyperchromatic cells, scant cytoplasm, tightly packed tubules, and glomeruloid-like structures  due to the finding of metastasis in the 7-year-old patient described previously . Such suIn this case, we treated a benign MA with robot-assisted laparoscopic partial nephrectomy. MA cannot be differentiated from other malignant neoplasms based on imaging. Renal biopsy is an option in the appropriate setting. If no histologic diagnosis is available, MA should be treated as all other renal masses with partial or radical nephrectomy, cryoablation, or radiofrequency ablation. MA is easily recognized microscopically and differentiated from other renal neoplasms. Though a benign disease, followup is appropriate with radiographic imaging including chest radiograph and computed tomography.


A 17-year-old Hispanic female presented to our hospital with complaints of right upper quadrant abdominal pain, vomiting, and fever. Physical exam was positive for hepatomegaly. Abdominal computed tomography showed multiple hypoechoic liver masses. Liver biopsy was done, which was diagnostic for hepatic epithelioid hemangioendothelioma.  Hepatic epitheliod hemangioendothelioma (HEH) is an extremely rare malignancy originating from vascular endothelial cells . HEH wasBelow, we present a case of a 17-year-old female who presented to the hospital with complaints of right upper quadrant abdominal pain. Abdominal imaging revealed multiple liver masses, and biopsy was diagnostic for HEH. We will briefly discuss the available literature on HEH.A 17-year-old Hispanic female presented to the emergency department with complaints of the right upper quadrant abdominal pain. The pain had worsened over the past three days. Initially the pain was intermittent and later progressed to be constant. Review of symptoms was positive for vomiting and fever. No change in bowel habits or urinary pattern was reported.On physical examination, the patient had right lower and upper quadrant tenderness with hepatomegaly two centimeters below the right costal margin. Laboratory tests including complete blood count, comprehensive metabolic panel, lipid panel, and pancreatic enzymes were within normal limits. Abdominal ultrasound was done, which showed multiple hypoechoic masses scattered throughout the liver, measuring up to 2.0 cm in size. Computed tomography (CT) scan showed multiple low attenuation areas in the liver . Liver bGiven the multifocal hepatic disease in this patient, she was not deemed to be a candidate for liver resection. The patient was referred for liver transplantation and is currently on the waiting list.HEH is a very rare liver tumor originating from vascular endothelial cells . The disMean age at presentation is 41.7 years according to a review of 434 cases  and may Unfortunately, no laboratory biomarker is available for screening. However, cancer biomarker CA19-9, if abnormal, may indicate a poor prognosis in patients with HEH . AbdominHEH has a highly variable prognosis ranging from a benign course to rapidly progressive with a poor outcome. Treatment is based on the radiological features of HEH: in a limited disease (less than three liver segments involved) tumor resection can be achieved, whereas liver transplantation is recommended for patient with multifocal liver disease . TranscaSeveral case studies reported that chemotherapy with radiation therapy may be useful in patient with HEH . Two recIn conclusion, HEH is a very uncommon disease with nonspecific clinical features. Imaging studies are of some utility, and hepatic biopsy is essential to establish a diagnosis. Given its extremely rare incidence, it is almost impossible to run randomized controlled trials, and the clinicians guide the management based on the published case series.


We report here a case of thymoma simultaneously associated with undifferentiated pleomorphic sarcoma. A 45-year-old male presented with axillary lump. Radiographic studies showed a mediastinal mass. On fine needle aspiration cytology and histopathological examination, a diagnosis of thymoma with coexisting undifferentiated pleomorphic sarcoma was made. Although thymomas are associated with many extrathymic malignancies, it's association with undifferentiated pleomorphic sarcoma is rare. This case is being reported on to reinforce that clinicians should bear in mind the possibility of extrathymic malignancies in patients with thymomas. Thymoma is a tumor of anterior mediastinum, however, thymomas may develop at various other sites like neck, trachea, thyroid, lung, and heart . It is tA 45-year-old male presented with easy fatigability and lump in the left side of axilla for four months. On general examination mild pallor was noticed. Local examination of left axilla showed a lump of 8 × 8 cm., firm in consistency, fixed to underlying structures, and overlying skin was normal . InvestiThe coexisting thymomas with other malignancies is relatively rare, and the most common site for these extrathymic malignancies is colorectal , 8 folloUndifferentiated pleomorphic sarcoma is the most common soft tissue sarcoma in adults, usually arising in extremities with a peak in seventh decade although cases in children have been reported , 16. It


This study focuses on CT and MR studies of adult patient with giant lesion of the posterior cranial fossa associated with micro- and macroaccumulations with density and signal like “fat” at the level of the cortical and cisternal cerebral spaces. This condition is compatible with previous asymptomatic ruptured dermoid cyst. Histological findings confirm the hypothesis formulated using the imaging. We also integrate elements of differential diagnosis by another giant lesion of the posterior cranial fossa. Dermoid cyst is a congenital dysembryogenic lesion representing only 1% of intracranial lesions. The posterior cranial fossa is one of the possible development sites, and cystic growth is stimulated by many factors like sebum secretions and/or scaling of internal cystic wall.Symptoms are related to dislocation and compression of brain parenchyma. The progressive lesion growth favours the rupture followed to cystic material dissemination in the cortical and cisternal brain spaces. In ruptured cyst the chemical meningitis can be subsequent. We present a case report of giant dermoid cyst on the posterior cranial fossa associated to micro- and macroaccumulation  widespread in the cortical and cisternal cerebral spaces. Elements of differential diagnosis from other giant lesion interesting the posterior cranial fossa have been integrated into the discussion.A 56-years-old woman was admitted at our neuroscience department complaining a recent slight diplopia and persistent headache which had arisen about five-six months before, initially NSAID responsive, that was progressively increasingly. A first medical examination showed peripheral right facial nerve and right abducent nerve paralysis. At the examination, vital signs and blood parameters were normal. No other neurological signs were detected.A CT brain scan was performed with reconstruction in 2D MPR and VR that showed a presence of a giant expansive lesion on the right side of posterior cranial fossa . The lesion developed from the posterior cranial fossa toward foramen magnum  till midA cerebellar vermis on the IV ventricle was displaced too  causing Volume rendering and MPR images showed massive erosion of the right petrous apex and great wing of sphenoid, followed the diameter increasing of right inferior orbital foramen.The densitometric difference evidenced during the examination showed an a cranial area iso-hypodense (−10/−5 HU) and a hypodense caudal area (−50/−60 HU) that seemed like a fat component. CT scan showed, moreover, a different marked hypodense lesions (−150/−180 HU) spreading over supratentorial cisternal and cortical spaces Figures . To reach a more comprehensive view about the lesion and its relationship with the intracranial circulation, an MR scan was performed.The FSE T2-weighted, FLAIR, EPI, 3D T1-fat suppression, and 3D T1-enhancement sequences were performed on the three orthogonal planes. Moreover evaluation of intracranial circulation was assessed trough the acquisition of 3D-TOF, used for post-processing images like 3D-MIP and 2D-MPR.The lesion caudal area appeared homogeneously hyperintense Figures  and heteThe dark signal in the lesion was suggestive for hemosiderin presence linked by previous bleeding Figures . After iThe other multiple hyperintense areas in T2-w and T1-w images (linkable with fatty droplets) appeared spreading to the supratentorial cisternal and cortical spaces ; after contrast administration no enhancement was showed. Figures . In 3D-TOF sequences, we have found abnormal course of the basilar artery dislocated and stretched toward the left side, the right posterior cerebral artery that was raised and lengthened and the right posterior communicant artery that appeared extremely stretched.The careful analysis of the acquired data provides the clue of the primary disorder: a giant dermoid cyst.The patient underwent partial resection of the lesion with transtemporal approach whose aim was to reduce mass effect on the brainstem and right temporal bone.During the surgery, many hair and some bone fragments escaped confirmed its origin.By the way material was collected to perform the histological investigation which, at the last, confirmed the dysembryogenic nature of the lesion. .Intracranial dermoid cyst is a congenital dysembryogenic lesion  that devBesides usually we do not find mesodermal elements and adipose tissue within this lesion.Lunardi et al. originate intracranial dermoid cyst from ectodermic nest cells dysembryogenetically adherent to primitive veins .It is a benign, ectopic, and squamous epithelial cyst containing dermal elements, including hair follicles, sebaceous, and sweat glands. This lesion accounts for 0,5%–1% of all intracranial lesion and is four to nine times less common than epidermoid cysts. Its development occurs between the 2nd and the 3rd decade of life and slightly prefers males. Sporadically, can be associated with Goldenhar  and Klippel-Feil syndromes. When the entrapment of embryonic ectoderm tissue into the neural tube occurs between the 5th and 6th week of fetal life, the lesion usually will be localized in midline; if the entrapment occur later, during the formation of secondary cerebral vesicles, it will be easily ahead from the midline [Our patient was presented with unilocular giant lesion starting from basal cisterns extending down towards the foramen magnum with great dislocation of the brainstem and the cerebellar vermis. In the cranial site, it was pushing the tentorium brain invading the right middle cranial fossa and causing compression of right temporal lobe. The pathophysiology of these is related to their progressive growth with consequent compression and dislocation of nearby neurovascular structures. Its growth is supported by endocrine influence that is increasing with age, in that case the dermoid cysts can secrete sebum and sweat, and, sometimes, exfoliating squamous epithelium can be found in the cyst. By that, this injury is often revealed only in adulthood, and its progressive growth is responsible of rupture and subsequent, possible, chemical meningitis.The most common symptoms are headache (32%) and seizure (30%) and when the cyst becomes particularly large, symptoms will be related to compression of neurovascular structures and eventually developing of cerebral hypertension syndrome. Chemical meningitis occurs in a high percentage of patients after rupture of dermoid cyst. The possibility of malignant degeneration in the sense of squamous cell carcinoma is rare but possible complication .On CT images, this lesion appears like a uniloculated, well-delineated, cystic mass with hypodensity from moderate to significant (from 0 to −150 HU), sometimes are present capsular calcifications. Generally, does not show enhancement after contrast administration.MR findings , 4 are hThe “fat” density or signal seen in the images is not due to the presence of adipose tissue but depends on the internal composition of the lesion like cholesterol. Heterogeneous aspect depends on a keratinaceous debris, sebaceous and sweat secretion as well as appendages including hair, and rarely nails or teeth.In our case it has been possible to detect the presence of multiple micro- and macroaccumulation in the cisternal and cortical cerebral spaces: this finding is typically compatible with previous asymptomatic rupture. The diffusion which is also linked to progressive growth of the lesion allowed to release cholesterol crystals in the subarachnoid spaces which resulting in widespread of lipid collected. This condition is frequently reported and well documented after rupture of intracranial dermoid and epidermoid cysts –7. RuptuWhen the cholesterol crystal is spread by cerebral spinal fluid (CSF) it tends to migrate into subarachnoid spaces particularly at the level of cerebellopontine angle, suprasellar and prepontine cistern and along the sagittal sinus. In our patient, massive dissemination was represented by macro and microcollect of fatty elements.Lipoid aseptic meningitis  is the mHistopathological result of the biopsy confirmed the neuroradiological suspect.In our case, erosion of the petrous apex and sphenoidal great wing with an increasing diameter of the inferior orbital foramen made opportunely a differential diagnosis with giant cholesteatoma and cholesterol granuloma . The chaThe most important differential diagnosis from other intracranial midline lesions includes epidermoid cyst, craniopharyngioma, teratoma, and lipoma.Epidermoid cyst  arises from normal epithelial cells included during neural tube closure; this lesion tends to become “giant” through a progressive accumulation of cholesterol and keratin due to the desquamation of the lining epithelium, like dermoid cyst. The imaging difference between them on CT scan is that epidermoid cyst presents hypodense with HU score similar to the CSF; also bone erosion is present. On MR scan shows often slightly hyperintense to CSF on T1-w images; less commonly is hyperintense to brain due to high contents of triglycerides and unsaturated fatty acids on T1-w . When solid crystal cholesterol and keratin predominate the lesion appears hypointense to CSF on T1-w . On T2-w images, it is often isointense or slightly hyperintense to CSF. On DWI, it is characteristic hyperintense; often, it is necessary to differentiate them from arachnoid cyst [oid cyst , and it The distinction between dermoid and epidermoid cysts is prognostically relevant and may impact on surgical management. In dermoid, the surgical choice is usually represented by subtotal resection also because it is rarely followed by lesion recurrence. In epidermoid, it prefers a total resection. Furthermore, the epidermoid cystic has a low incidence of rupture also because its growth is not regulated by hormonal factors but only by squamous exfoliation of inning layer.teratoma, as well as being possible through the “Imaging,” is that it take origin from all three germinal layers  [The difference from the ndoderm) , 4, instTeratoma has also extremely heterogeneous signal: iso-hyperintense both T1-w and T2-w due to the presence of calcification, CSF, fat, and soft tissue components; typically, teratoma may contain adipocytes (to the opposite of dermoid). On DWI, the solid component which shows a restricted diffusion after contrast administration enhancement of the soft tissue is typical.lipoma is a well-delineated mass of mature nonneoplastic adipose tissue often associated with agenesis of the corpus callosum; it is usually more homogeneous than dermoid cyst and capsular calcification has rarely. Lipoma shows marked hypodense on CT scan (−50 to −100 HU); on MR scan, it is hyperintense on T1-w and hypointense on T2-w and fat suppression, without contrast enhancement.Intracranial Craniopharyngioma is an aggressive lesion often found in midline including the intra- and suprasellar region with serious skull-based erosion, frequently finding on CT images. In childhoods (adamantinomatous type), usually presents a solid (isodense) mass whit cystic (hypodense) component (sometimes multi cystic); nodular calcifications and nodular or rim enhancement are frequent findings. In adults (papillary type), lesion shows isodense with nodular enhancement.Common MR findings for the solid component are heterogeneous signal on T1-w and T2-w images with heterogeneous enhancement; cystic components are variably hyperintense on T2-w while signal varies with the cyst contents on DWI and T1-w images (short T1 due to high protein content), after contrast cyst wall shows strongly enhancement. Dermoid cyst showed minimal or no enhancement. This lesion is more common than dermoid cyst, and normally it is easily to differentiate from other intracranial lesions in the midline. CT and MR images have been relevant for correct diagnosis that allows the most appropriate choosing for a better surgical procedure case by case. The most frequently selected surgical approaches are the middle cranial fossa or, if possible, transtemporal that provides the widest surgical view preserving acuity of hearing. Our patient has been undertaken to transtemporal approach with complete excision of the lesion, but, in absence of previous medical history of meningitis, it was not considered necessary to remove the lipid macrocollected.Total removal is not recommended when these lesions adhere to the neurovascular structures as this is associated with high morbidity and mortality; in our case, the lesion showed a surgical cleavage plane without adhesions with the neurovascular structures of the middle and posterior fossa. The possibility of recurrence should, therefore, be considered especially when the capsule is not completely removed. Two weeks after intervention, our patient showed complete remission of the symptoms.Subsequent CT and MRI followup showed normal postsurgery course.


Rhabdomyosarcoma is a solid tumor, resulting from dysregulation of the skeletal myogenesis program. For rhabdomyosarcomas (RMS) with a predilection for the head and neck, genitourinary tract, extremities, trunk, retroperitoneum, the larynx is still an unusual site. Till now only several cases of this laryngeal tumor have been described in world literature in the adult population. The entire spectrum of genetic factors underlying RMS development and progression is unclear until today. Multiple signaling pathways seem to be involved in ERMS development and progression.In this paper we report an interesting RMS case in which the disease was located within the glottic region. We report an embryonal rhabdomyosarcoma of the larynx in 33 year-old man. After unsuccessful chemotherapy hemilaryngectomy was performed. In follow up CT no signs of recurrence were found. Recently patient is recurrence free for 62 months.Considering the histological diagnosis and the highly aggressive nature of the lesion for optimal diagnosis positron electron tomography (PET) and computerized tomography (CT) of the neck and thorax should be performed. At this time surgical treatment with adjuvant radiotherapy seems to be the treatment of choice for this disease. Rhabdomyosarcoma of the larynx has a better prognosis than elsewhere in the body, probably because of its earlier recognition and accessibility to radical surgery. More than 95% of laryngeal tumors in adults are squamous cell carcinoma. Laryngeal rhabdomyosarcomas are very rare, but extremely malignant tumors. Rhabdomyosarcoma must certainly rank close to, if not the least common of the laryngeal sarcomas. Most of reported cases have occurred in children . RhabdomAt the same time for oncology and surgery specialists the precise pre-therapeutical staging of tumors of the musculoskeletal system provides important prognostic information and has impact on the entire therapy management. During therapy imaging is extremely important in the follow-up and in diagnosing a possible recurrent disease . RecentlTill now only several cases of this laryngeal tumor have been described in world literature in the adult population -11. AlthRhabdomyosarcoma is a solid tumor, resulting from dysregulation of the skeletal myogenesis program. Four histological subtypes occur in RMS-embryonal (ERMS), alveolar (ARMS), pleomorphic and botryoid. The entire spectrum of genetic factors underlying RMS development and progression is unclear until today. While chromosomal rearrangements account for the majority of alveolar tumors, the genetic defects underlying the pathogenesis of embryonal type remain largely undetermined. Cytogenetically ERMS karyotype is disturbed, most frequently hyperdiploid, with extra copies of chromosomes 2, 7, 8, 11, 12, 13, and 20. Aneuploidies typical of sporadic E-RMS include gain of chromosomes 3, 8, 13 and loss of chromosomes 9, 14, X , loss ofMolecular genetics analyses have not only assisted in understanding the molecular mechanism in sarcoma pathogenesis but also demonstrated new relationships within different types of sarcomas leading to a more proper classification of sarcomas .Multiple signaling pathways seem to be involved in ERMS development and progression. Array-based CGH analysis in primary RMS detected copy number changes of transcription factors such as MYC-related and the cytoskeleton and cell adhesion gene-laminin gamma-2 and also p21-activated kinase-1. In the In the majority of ERMS cases screened, overexpression of MCL1 (induced myeloid leukemia cell differentiation protein) and MAP2K4 (mitogen-activated protein kinase 4) genes, both involved in cell viability regulation, was reported . At the Moreover FGFR1 (fibroblast growth factor receptor 1) overexpression was detected in all primary RMS tumors and cell lines tested. A hypomethylation of a CpG island upstream to FGFR1 exon 1 was identified, which suggests a molecular - epigenetic - mechanism of FGFR1 overexpression . Recent Not only proteins but also microRNAs (miRNA) have been implicated in RMS development and the clinical behavior of RMS. Muscle-specific miRNAs levels are lower in RMS compared with skeletal muscle but generally higher than in other normal tissues. Low miR-206 expression correlates with poor overall survival and is an independent predictor of shorter survival in metastatic embryonal RMS cases without PAX3/7-FOXO1 fusion genes. Low miR-206 expression also significantly correlates with high SIOP stage and the presence of metastases at diagnosis. High miR-206 expression strongly correlated with expression of genes involved in muscle differentiation and low expression is associated with genes linked to MAPkinase and NFKappaB pathway activation . At the All data collected till now suggest the involvement of genes encoding cell adhesion, cytoskeletal signaling as well as transcriptional and cell cycle components in RMS tumorigenesis . It is n33 years old male patient was referred to Department of Otolaryngology, Medical University of Warsaw, due to isolated longstanding hoarseness. At first visit weight was 98.5 kg, and height: 186 cm. The patient had no medical history (diagnoses or operations), was non-smoker and was not under any treatment.rhabdomyosarcoma embryonale. For treatment planning in August 2005, computed tomography (CT) examination of the larynx was performed with contiguous 2.7 mm thick slices and showed a poorly delineated tumor located on the right side of the larynx, invading the vocal fold and the ventricular fold, contracting the pyriform recess and constricting the glottis. Contrast enhancement in the tumor was observed in the CT scans . The first chemotherapy cycle began on September 8st 2006. Chemotherapy was continued until August 24th 2006. In total the patient received twelve cycles of VAC1, with 50% dose reduction after V cycle due to neutropenic fever in November 2005. The patient had the nasogastric tube removed in May 2006 and was decaniulated in October 2006. Following the surgery, the patient was monitored by a laryngologist and oncologist and underwent regular fiberoptic examinations of the larynx and direct laryngoscopy in case of unclear fiberoptic examination results. Follow-up CTs were performed in October 2006, June 2007, November 2007, June 2008 and March 2009, and did not reveal any tumor recurrence .AMC, WK-have been involved in drafting the manuscript, AK, WK, PW, BJ, GS-diagnosed and treated the patient, AMC, WK, AK-have made substantial contributions to the conception and design of the manuscript. All authors have read and approved the manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2407/11/166/prepub


To report a case of non-Hodgkin’s lymphoma (NHL) presenting as an ocular adnexal and forehead mass.An elderly male patient was referred by a neurosurgeon to the eye clinic with a six-month history of a massive tumor measuring 12×16×8 cm involving the right side of the forehead, eyebrow and upper eyelid. Neurological examination had been normal and computed tomography revealed no intracranial extension. The patient was referred to an otorhinolaryngologist who performed an incisional biopsy which revealed the mass to be NHL. He received chemotherapy with CHOP regimen  resulting in reduction in lesion size leaving a phthysical eyeball and a ptotic lid.Non-Hodgkin’s lymphoma may occur in almost any part of the body and should be considered in the differential diagnosis of extralymphoid tumors. Non-Hodgkin’s lymphoma (NHL) comprises 90% of all hymphomas.4Lymphoma may present as a localized eyelid mass or nodule.A 63-year-old male subject, a retired trader, presented to the University of Benin Teaching Hospital, Benin City, Nigeria, with a mass lesion involving the right side of the face of 6 months’ duration. There was no history of trauma, but the patient had experienced weight loss.He had first presented to a general hospital from where he was referred to a neurosurgery center to rule out intracranial lesions. He underwent cranial computed tomography (CT) scanning which showed that the mass had no intracranial connection or extension and that the eyeball was intact . He was On ophthalmologic examination, the patient was not in distress, but the mass was very large involving the right side of the forehead, right eyebrow and the right upper eyelid such that it covered the cheek and extended down to the nostrils. The right eyeball could not be visualized . The masHistology of the biopsy specimen disclosed sections of neoplastic tissue made up of small lymphocytes with mildly pleomorphic vesicular nuclei, arranged diffusely; most had plasmacytoid nuclei . A diagn2 IV, days 1 and 8; adriamycin 45 mg/m2 IV, days 1 and 8; vincristine 1.5 mg/m2 IV, days 1 and 8 and prednisolone 20 mg PO, TDS). The mass was considerably reduced in size after the second course of chemotherapy  in the frontal region which was noticed following head injury. Six months earlier, he had declined treatment for malignant lymphoma , discovered upon evaluation of a subcutaneous mass in his forearm. Computed tomography disclosed a large, homogeneously enhancing mass with both extradural and extracranial extension which had destroyed the left frontal bone. The skull tumor was completely resected and a diagnosis of malignant lymphoma, large-cell, immunoblastic and plasmacytoid type, was confirmed.7The third case was a 65-year-old male subject who complained of a huge and rapidly growing scalp mass (largest dimensions 13×9×4 cm) and hemiparesis. The craniogram revealed a large osteolytic lesion in the right parietal bone. The histological diagnosis was malignant lymphoma (B cell type).8Although there are a few other reports of lymphoma affecting the skull, only the first two of the abovementioned cases, presented in the frontal area but were not large enough to affect the upper lid and cover the cheek. The current report shows that NHL may present at unusual sites and pose a diagnostic dilemma. Such cases can cause delays in initiating definitive treatment with potentially disastrous consequences for the patient.


A villous adenoma is an extremely rare benign tumour in the appendix, in contrast to other benign appendiceal lesions. The clinical features are usually asymptomatic. Acute appendicitis is the most common complication with the lesion obstructing the orifice of the appendiceal lumen. Thus, a villous adenoma is usually found during surgical intervention for acute appendicitis. Mechanical obstruction induced by acute perforated appendicitis has been previously reported. Acute appendicitis caused by a villous adenoma presenting with acute intestinal obstruction has not been previously reported.A 78-year-old woman presented to our Emergency Department with diffuse abdominal pain and tenderness. The abdominal plain film and computed tomography revealed an intestinal obstruction. After surgical intervention, the ruptured appendix was shown to be associated with intestinal obstruction. The post-operative pathologic diagnosis was an appendiceal villous adenoma.This is the first report describing an appendiceal villous adenoma, which is an occasional cause of perforated acute appendicitis, presenting as a complete intestinal obstruction. We emphasize that in elderly patients without a surgical history, the occult cause of complete intestinal obstruction must be determined. If an appendiceal tumour is diagnosed, an intra-operative frozen section is suggested prior to selecting a suitable method of surgical intervention. Intestinal obstruction is a common problem in clinical practice. The common causes of intestinal obstruction are mechanical obstruction or ileus. Although post-operative adhesions account for up to 75% of small bowel obstructions, it is notable that acute appendicitis is a rare cause of small bowel obstruction ,2. AcuteClearly, appropriate surgical intervention is very important. Herein we present a case of complete small intestinal obstruction caused by acute appendicitis with perforation resulting from obstruction by a villous adenoma.3/μL (95% neutrophils) and a C-reactive protein (CRP) level of 35.20 mg/dL . An abdomen plain film showed dilated bowel loops in the left abdomen  complicated with intestinal obstruction . VillousIn our patient, no previous presentations of acute appendicitis, such as local tenderness, local rigidity over the right lower quadrant abdomen, or McBurney's point tenderness were found on the initial examination, for the following reasons.First, the peritoneum contained the perforated appendix within the sac, preventing widespread inflammation. Thus, in this patient the presentation was limited to local tenderness, and no muscle guarding or peritoneal signs were noted. Second, the co-existence of the two diseases might change the typical pathogenesis of this condition. In general, the sudden onset of complete small bowel obstruction with adhesion band formation in patients without a surgical history must be carefully investigated to identify the underlying problems of this simple disease.The current opinion regarding the exclusion of malignant lesions depends on whether or not the resection line of the appendiceal stump is clear. Appendiceal villous adenomas have a propensity to develop into invasive adenocarcinomas, such as adenomas, elsewhere in the colon. Thus, an intra-operative frozen section is suggested. Appendectomy is the treatment of choice for those cases in which the lesion does not reach the line of resection . Once maThis is the first report of an unusual presentation of perforated appendicitis induced by an intraluminal villous adenoma. No previous report of a similar presentation  exists in the literature. We emphasized two key points. First, acute appendicitis and complete intestinal obstruction are surgical emergencies. However, in the elderly with a complete mechanical intestinal obstruction without a previous surgical history or malignancy, the possibility of occult reasons or a co-existing disease, such as acute appendicitis or hernias, must be considered. Second, an appendiceal villous tumour must be considered as a possible cause of acute appendicitis. A post-operative colonoscopy should be considered to check for synchronous or metachronous colon cancer.The authors declare that they have no competing interests.YGC and YLC was involved in writing, reviewing, editing and finalizing the manuscript. HMC and YCC managed the whole operative procedures. YGC, YCC and HMC participated in the care of the patient and assisted in drafting the manuscript. CHH was the specialist in the gastroenterology involved with the case and also reviewed, edited and finalized the manuscript. All authors have read and approved the final version.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-230X/11/35/prepub


The abdominal cocoon syndrome (or idiopathic encapsulating peritonitis) is a rare cause of intestinal obstruction. It has been reported predominantly in adolescent girls living in tropical/subtropical region in which diagnosis is only made at laparotomy in most cases. The cause and pathogenesis of the condition have not been elucidated. Prolonged administration of practalol, meconium peritonitis, and tuberculous infection of the female genital tract have been incriminated as possible causes. The author reports a case of a female patient with recurrent intestinal obstruction treated for years but failed to settle down on conservative treatment during her last hospital admission and had to undergo surgery. Preoperative diagnosis of this syndrome as the cause of her intestinal obstruction was not made until at laparotomy, when a thick fibrotic peritoneal wrapping of the bowel in a concertina-like fashion with some adhesions was found. Excision of this membrane and adhesiolysis were carried out without any need for bowel resection, and this led to relief of the obstruction and patient's complete recovery. Awareness of this benign condition in the differential diagnosis of intestinal obstruction will result in early diagnosis and correct management and prevent unnecessary bowel resections and bad outcomes. Abdominal cocoon syndrome (or idiopathic encapsulating peritonitis) is a relatively rare cause of intestinal obstruction, described mostly in young adolescent girls, and was first described in 1978 [The etiology of this disease is largely unknown and the cases seen so far have been limited to the tropical and subtropical zones and primarily affect young adolescent females, even though several earlier cases have been reported also in males , 3.Thick fibrotic peritoneum encasing the small bowel partially or completely is a pathognomonic feature, and the correct diagnosis is not often made preoperatively.This is the case report of an eighteen- (18-) year-old Nigerian girl that presented with intestinal obstruction  that faiThe author is therefore presenting the case so that practicing surgeons have better awareness of this condition as a possible cause in the differential diagnosis of intestinal obstruction which may facilitate pre-operative diagnosis, prevent inadvertent bowel damage at laparoscopy, and prevent unnecessary bowel resection at laparotomy .An eighteen- (18-) year-old Nigerian girl presented with severe central/lower abdominal pain associated with a few episodes of bilious vomiting which started about two (2) to three (3) days at home before admission to the hospital.She took some self-medication at home which is a common practice in this part of the developing world, but unfortunately her condition did not improve.Clinical examination revealed a moderately ill-looking slightly dehydrated young lady. Temperature 37.8°C; BP 100/70 mmHg; Pulse 80/min; RR 24/min. Abdomen was tender and moderately distended at its central/lower part.A palpable mass at the right lower paraumbilical area was found. Bowel sounds were found to be sluggish.Significant in her past medical is the fact that she had recurrent antibiotic therapy for the past three years following an undisclosed intravaginal medical treatment by a local quack practitioner.Plain abdominal X-rays were unremarkable  month.Abdominal cocoon syndrome (or idiopathic encapsulating peritonitis) is a rare disease of the peritoneum and almost invariably presents as an acute or subacute intestinal obstruction with or without a mass, which is usually diagnosed incidentally at laparotomy .It was first described by Foo et al. in 1978 [Some authors have implicated prolonged administration of practalol therapy as a possible aetiology in some cases, meconium peritonitis, sarcoidosis, orthotopic liver transplantation, indwelling abdominal catheters, or even tuberculous infection of the female genital tract .These conditions may predispose patients to chronic peritoneal irritation and inflammation, which as a final effect leads to peritoneal fibroneogenesis.Cell-mediated immunological tissue damage initiated by microorganisms assessed by immune-fluorescent studies has been documented in some cases as possible cause. Pre-operative tissue culture of peritoneal membrane may also contribute in future in the further evaluation of the aetiology of abdominal cocoon syndrome . In this patient, it was probably secondary to her previous intravaginal treatment a couple of years prior to her presenting with recurrent lower abdominal pain and subacute intestinal obstruction. To the best of the author's knowledge, this the second recorded case in Nigeria . She couDespite anecdotal reports of a preoperative diagnosis especially by medical imaging, in the majority of cases of idiopathic encapsulating peritonitis, it is a fortuitous finding. A better awareness of this condition is what may facilitate pre-operative diagnosis , 12.A high index of clinical suspicion , 14, witClinicians must rigorously pursue a preoperative diagnosis, as it may prevent a “surprise” upon laparotomy , 18 and


Intramuscular myxoma (IM) is a benign intramuscular neoplasm composed of fibroblasts and abundant myxoid stroma. IMs most commonly affect larger skeletal muscles, while those affecting the oral and maxillofacial regions are rare, with a small number of documented cases in the available literature. The aim of the present study was to describe a highly rare case of an IM within the hyoglossus muscle of the tongue in a 74-year-old male. The patient presented with a painless mass in the submental space that had been growing slowly for more than five years. A computed tomography scan revealed a hypodense lesion located in the root of the tongue. The mass was easily excised with thin margins, including only a small amount of the adjacent muscle tissue. The pathological diagnosis of the mass was an IM. The patient made an excellent recovery following the surgery and the follow-up three years later revealed no local recurrence. IMs of the hyoglossus muscle are highly rare, however must be considered in the differential diagnosis of swellings in the root of the tongue region. Myxoma is a benign tumor of mesenchymal origin composed of undifferentiated stellate cells in a loose mucoid stroma with delicate reticulin fibers. The diagnostic criteria of the myxomas were initially proposed by Stout . These tA 74-year-old male patient presented to the First Affiliated Hospital of Yangtze University  with a painless mass in the anterior region of the upper neck that had been growing slowly for more than five years; the patient noted that the expansive mass had exhibited a progressive volume increase within the last six months. The patient also complained that swallowing food and pronunciation had been affected by the mass. The patient had no history of trauma, fever or weight loss. Clinical examination revealed a firm and tender mass  in the root of the tongue.Computed tomography (CT) of the neck revealed a hypodense lesion located in the anterior cervical region of the neck, on the hyoid bone within the hyoglossus muscle. The CT value of the tumor was ~20.3 HU (Hounsfield unit). The oropharyngeal cavity became narrow as a result of tumor pressure  and the Surgical resection was accomplished through a horizontal incision on the hyoid bone over the tumor site, under general anesthetic. The tumor was easily separated from the normal muscle margins of the root of the tongue and the mucous membrane of the root of the tongue, which was adhered to the tumor was resected.The tumor was encapsulated intact and macroscopically, it exhibited a gray-white appearance and the cut surface of the specimen was ovoid, yellow and gelatinous . HistopaIn 1871, Virchow  used theThe clinical diagnosis of IM is problematic prior to microscopic examination due to the oral and maxillofacial location, as IM exhibits non-specific clinical manifestations. Therefore, the clinical differential diagnosis includes benign tumors of mesenchymal origin, for example, benign muscle neoplasms, such as rhabdomyoma and leiomyoma. In the present case, the patient’s tumor was located in the root of tongue, therefore various diagnoses could have been considered, including a more common benign tumor or cyst of the tongue, such as a dermoid or epidermoid cyst, teratoma or neurilemmoma. The mass may also have been a rare benign tumor, such as a tumor arising from the lingual ectopic thyroid or lipoma, or it may have been a benign salivary gland tumor (pleomorphic and monomorphic adenoma) arising from the small salivary glands of the root of the tongue.The majority of tumors are solitary, however, a small proportion are multiple and are associated with fibrous dysplasia. The combination of multiple tumors, or more rarely solitary IMs, with skeletal fibrous dysplasia in now termed Mazabraud’s syndrome ; when anIMs lack specific radiographic features and CT scans typically reveal a cystic-like mass, with a CT value that is greater than water, but less than the surrounding normal muscle . These rDue to a lack of specific symptoms and common laboratory tests for identifying IMs, the diagnosis of IMs is difficult. It is very rare for these tumors to be correctly diagnosed prior to biopsy and histological examination. It has been reported that carbohydrate antigen (CA) 19-9, a tumor marker, may be correlated with IM. In a previous study, the serum level of CA 19-9 increased preoperatively and returned to a normal level six months following surgery , howeverMacroscopic analysis of IMs has demonstrated that the majority of these tumors are ovoid or globular, and covered by bundles of skeletal muscle, with a yellow/gray and gelatinous cut surface. Microscopic visualization using hematoxylin and eosin staining, shows that IMs are hypocellular, hypovascular, intensely mucoid and basophilic, and are composed of stellate and spindle-shaped cells in a myxoid stroma; certain IMs exhibit focal areas of hypercellularity and hypervascularity ,21. HypeIMs present as benign masses and there have been no reports of cases involving metastasis or other malignant changes. As a result, the treatment for IM is surgical excision, including enucleation, simple excision and wide local excision. Previous studies demonstrated several cases of recurrence following incomplete excision or simple enucleation due to the adjacent muscle tissue being infiltrated or an incomplete capsule ,23,24. HIn conclusion, the occurrence of IM in the hyoglossus muscle is highly rare. To the best of our knowledge, this is the first study of this type of tumor in this region. An accurate diagnosis prior to surgery is difficult due to a lack of characteristic clinical history and radiographic findings; therefore, a CT scan or MRI is required for treatment planning. Furthermore, IM must be considered in the differential diagnosis of swellings in the root of the tongue and simple excision was identified as a feasible method for the treatment of solitary IM.


Calcifying cystic odontogenic tumor (CCOT) demonstrates considerable diversity in histopathology and clinical behavior. Ghost cell odontogenic carcinoma (GCOC) is the rare malignant counterpart of CCOT and it frequently arises from malignant transformation of a recurrent CCOT. In this paper, we present a case of CCOT and discuss its distinct histopathologic features in recurrence. Then, we will have a review on clinical, histopathological, and immunohistochemical aspects of GCOC in the literature. Predictive factors of malignant transformation in a benign CCOT will also be discussed. Calcifying odontogenic cyst (COC) represents 2% of all odontogenic lesions in the jaw . It demoA 54-year-old male presented with swelling in the right side of mandible. He had a history of right first molar extraction 5 years ago with subsequent abscess formation and without any treatment. Radiographic examination revealed a multilocular radiolucent lesion . Root reThe primary and recurrent cases went through microscopic evaluation with immunohistochemistry including p53 and Ki-67. P53 staining was negative in both cases whereas Ki-67 labeling index was increased in the recurrent case with a mean of 5% in cribriform epithelial nests, confirming the proliferative activity of recurrent case Figures  and 9. TGCOC is the rare malignant counterpart of CCOT and approximately, 30 cases have been reported in the literature. GCOC is diagnosed on the basis of atypical histologic features, necrosis, prominent mitoses, infiltrative growth pattern, aggressive behavior, and high expression of Ki-67 and p53 . This maCCOT has no distinct predilection to maxilla or mandible and is slightly more common in women . In contWe observed increased Ki-67 labeling index in cribriform nests of recurrent case. However, p53 expression was rarely seen in tumor cells. Some authors have performed immunohistochemical examinations in CCOT and GCOC as its malignant counterpart. To conclude, malignant transformation should be considered in all recurrent CCOTs particularly in maxillary lesions and male patients . MoreoveCCOT is a benign lesion and most surgeons enucleate the lesion and curette 1-2 mm of the surrounding bone to remove any tumor remnant . HoweverMalignant transformation can occur in CCOT rapidly or after a long time. Arashiyama et al. reported a case of calcifying odontogenic cyst that transformed to malignancy after eighteen years . Therefo


Although small bowel intussusception is one of the most common abdominal emergencies in childhood, it is rare in adults and usually occurs as a result of an underlying pathology. Sarcomatoid carcinoma, a very rare subtype of lung cancer, rarely metastasizes to small bowel and causes complications. In this paper, we aim to describe a patient with small bowel intussusception caused by an isolated small bowel metastasis of the sarcomatoid carcinoma of the lung by reviewing the literature. Bowel obstruction is a major cause of morbidity and financial expenditure in hospitals around the world and is frequently encountered in emergency departments. It can develop for a variety of etiological reasons, with adhesions being the most common cause. Intussusceptions constitute 1–5% of all cases of bowel obstructions. Despite intussusception being commonly observed in childhood, it is a rare cause of bowel obstruction in adult populations. Small bowel intussusception (SBI) in adults can be observed in association with idiopathic or benign causes, or they may develop secondary to malignant pathologies. In the latter case, the underlying causes of intussusception can be primary small intestine tumors, such as lymphoma, gastrointestinal stromal tumors and carcinoid tumors, or the metastasis of cancers to the small intestine from other organs [A 71-year-old man was admitted to our emergency department complaining of abdominal pain and vomiting. He described the absence of gas and feces for two days. Physical examination demonstrated a distended abdomen and mild epigastric tenderness. Auscultation was indicated by hyperactive bowel sounds and a plain abdominal film showed gas-fluid levels. Aside from a hemoglobin value of 6 and a hematocrit value of 21, laboratory evaluations revealed no pathological characteristics. Based on these observations, the patient was monitored with a diagnosis of anemia and mechanical intestinal obstruction. It was also learnt that the patient had undergone surgery one year previously for a lung tumor, and that he had been diagnosed with sarcomatoid carcinoma of the lung based on histopathological evaluations . The patient had had no further complaints in the following one year. Abdominal computerized tomography (CT) identified a mass located in the small intestines . FollowiDespite being the most common abdominal emergency in early childhood, intussusception is a rarely encountered pathology among adults. Starting from a mass or a lead point in the intestinal lumen, it is formed by telescopic invagination of the proximal loop into the distal loop. While intussusception in childhood is frequently idiopathic, in adults it commonly develops as a result of an underlying benign or malign etiology . In the Lung cancer is the leading cause of cancer-related death worldwide. Lung carcinoma is capable of metastasis to other regions within the body. Patients are rarely symptomatic and can be diagnosed only following the development of complications such as obstruction, perforation, or hemorrhage. The proper evaluation of abdominal pathologies during follow-up of lung cancer is thus important. Despite Garwood et al. reporting on a series of 98 patients with lung cancer, with findings of gastrointestinal metastasis for adenocarcinoma (23.7%), squamous cell carcinoma (22.7%), large cell carcinoma 20.6%), and small cell carcinoma (19.6%), cases of intussusception associated with sarcomatoid carcinoma were very rare . This pa%, and smAlthough it is known that lung carcinoma can metastasize to the small intestine via the hematogenous or lymphogenous paths, the exact route followed during metastasis is not known. However, retrograde metastasis to the lymphatic system of the small intestines via the thoracic duct appears possible. Immunohistochemical staining during histopathological evaluation is important for proper identification. In a review regarding sarcomatoid of the small bowel, strong positive results were observed with vimentin, cytokeratin AE1/AE3, and CK7, while negative staining was observed for the S100 protein, muscle cell actin, desmin, carsinoembryonic antigen, CK20, and CK117, which are important in differential diagnosis from other malignant pathologies [The treatment plan for adult intussusception consists of segmental resection and primary restoration of continuity of the gastrointestinal tract. As malignant pathologies are frequently observed in cases of colonic intussusception, reduction is not recommended for these cases . HoweverIn conclusion, it is important to consider that intestinal metastases of lung cancers can occur regardless of the type of pathology, that these metastases may have different clinical manifestations, and that the diagnostic and therapeutic approaches should be planned accordingly. Therefore, the existence of lung malignancies in the history of any patient presenting with abdominal symptoms should be evaluated carefully.


Mesenchymal type tumors originated in the submucosa represent a small percentage of colorectal polyps. This is particularly true for polyps composed of more than one mesenchymal tissue type. We herein present the pathological features of an unusual polypoid proliferation of mature fatty, fibrous, and vascular tissues including vessels of diverse nature and size. The histological findings support a hamartomatous rather than a true neoplastic origin for this rare lesion. While most colonic polyps are of epithelial nature and originated in the mucosa, a small subset of them represents submucosal proliferations of mesenchymal type cells. The most common types include lipomas, leiomyomas, benign neural and vascular lesions, and gastrointestinal stromal tumors (GISTs) . In the A 66-year-old male with history of hepatitis caused by HCV presented with abdominal pain of 3-month duration that was not associated with meals or a specific time of the day. The patient had no constipation, diarrhea, or bloody stool. Physical examination revealed no abdominal mass or tenderness. Stool examination was negative for occult blood. The hemoglobin and serum carcinoembryonic antigen levels were within normal limits. A full colonoscopy revealed a smooth-surfaced and sessile round polyp in the cecum measuring 1.0 cm in diameter. The lesion, which was covered by normal-appearing mucosa, was excised using a cold snare. The patient is alive and well with no additional colorectal lesions two years after the polyp's resection.Macroscopic examination of the excised specimen showed a circumscribed round polyp, 1.0 cm in diameter, of soft consistency, covered by normal-looking mucosa. Formalin-fixed, paraffin-embedded tissue sections were stained with hematoxylin and eosin (HE) and Weigert's stain for elastic fibers and assessed immunohistochemically for the following markers: CD31 , D2-40/podoplanin , smooth muscle actin (SMA) , muscle specific actin (MSA) , desmin , and HMB-45 . Histologic sections revealed a submucosal proliferation of mature adipose tissue with fibrous septa containing numerous vessels of varied nature  lined by a benign endothelial lining Figures . WeigertSince colonoscopy was recognized as the gold standard screening procedure for colorectal cancer, polypoid mesenchymal lesions of the colon are increasingly being detected . In this Other entities that may be considered in the differential diagnosis include tumors of adipose and/or vascular tissues of the gastrointestinal tract. Angiolipomas, an important consideration in our case, have been occasionally reported in the gastrointestinal tract. We found 11 cases including one in the esophagus , three iLess probably to be confused with our case are lesions composed exclusively of adipose or vascular tissue. In fact, the most common lipomatous neoplasms in the gastrointestinal tract are lipomas and their most common location is the right colon. They usually develop as a small, mucosa-covered intramural polypoid lesions . Unlike In summary, we have reported a rare case of submucosal angiolipofibroma of the colon that we believe to be the first reported case in the colorectum in the English literature. This benign lesion appears to be of hamartomatous origin and may be treated successfully by standard polypectomy techniques.


The formation of a Hairy Polyp on the dorsum of the tongue is a rare condition that may hinder vital functions such as swallowing and breathing due to mechanical obstruction. The authors present the situation on a child with an approach of significant academic value.Imaging diagnostics with the application of a topical oral radiocontrastant was used to determine the extent of the tumor. Performed treatment was complete excision and diagnostics was confirmed with anatomopathological analysis.The patient was controlled for five months and, showing no signs of relapse, was considered free from the lesion.Accurate diagnostics of such a lesion must be performed in depth so that proper surgical treatment may be performed. The imaging method proposed has permitted the visualization of the tumoral insertion and volume, as well as the comprehension of its threatening dynamics. Although the Hairy Polyp, also called Teratoid Polyp, has its origins and classification hugely debated -3, authoHairy Polyp is more frequently seen among women, mainly newborns, scarcely happening to adults . Its preFor diagnostic purposes, imaging exams such as computed tomography (CT) or magnetic resonance (MRI) might be performed. Particularly we indicated the computed tomography with a topic radiocontrast as it may not only detected the presence, but also the dynamics of a tumor located on the dorsum of the tongue. This paper, therefore, aims to describe and discuss the presence of a Hairy Polyp in the tongue of a newborn girl, rapidly developing into complications regarding the vital functions of swallowing and breathing.A 90 days old female patient, according to information given by the mother, had shown progressive signs of difficulty in swallowing and breathing, together with repeated incidences of cough and vomiting. The constant refusal to suck the pacifier and to be breastfed, meant that it took a long time to feed the baby. The medical report informed that there was a delay in weight gain. At the first medical examination, a voluminous and mobile tumoral alteration was noticed on the dorsum of the tongue, resting anteriorly, but it wasn’t possible to stipulate whether it extended to the oropharyngeal area.A new exam was set and when the computer images were obtained – for which the patient was sedated under spontaneous breathing – a pediculated mass with a cylindrical body could be seen . It was located longitudinally on the dorsum of the tongue, with its base placed in the median furrow, at an almost equal distance between the apex of the tongue and the foramen cecum. The lesion was mobile to any direction, composed of a soft consistency, yielding to traction and with color and texture similar to those of the oral mucous membranes Figure .Computed topography images were obtained. Through the use of a non ionic oral radiocontrastant a polipoid lesion was clearly distinguishable on the dorsum of the tongue, measuring 2,1cm x 0,8cm. The use of an oral radiocontrast allowed not only for the delimitation of the lesion's contours, but also, its dynamics, demonstrating its movement from the dorsum of the tongue to the oropharyngeal area, suspending the soft palate (Figure Surgery was performed under general anesthetic, using nasotracheal intubation. A complete resection of the pedicle was performed, involving, in depth, the fibers of the superior longitudinal muscle of the tongue. After the hemostasis, the procedure was concluded with layer sutures to isolated points. The surgical specimen was stored in a 10% buffered formaldehyde solution and sent for anatomopathological analysis Figure .On the macroscopic examination a polypoid fragment of light brown tissue covered by a grayish and wrinkled mucous membrane was observed, measuring 0,8 x 0,9 x 0,8 cm. At the sections, in turn, it shows a bright light brown color.The microscopic examination revealed a polipoid lesion lined with keratinizing squamous epithelium, containing stroma, besides connective and adipose tissue, smooth and striated muscle and salivary glands Figure - a,b,c,dThe postoperative period evolved without complications. In the five-month follow up there wasn’t any recurrence and the patient, therefore, was considered free from the lesion.In spite of its uncommonness, the Hairy Polyp is recognized as a significant pathology that affects both naso and oropharynx. It’s a congenital tumor quite usual for those areas . HoweverDisplaying an approximate incidence of 1 per 40.000 births, most of the cases are considered isolated. According to Burns, Axon and Pahade , being eHairy Polyp as accurate and explanatory, for the process is considered as a developmental malformation. Its histology, though variable, is typical, and likely to be distinguished from other lesions that may affect the patient, dissociating itself from malign precesses. On the contrary to the epignathus, teratomas and trigeminal teratoids, the Hairy Polyp doesn’t have the endodermal layer in its origin [Carranza  regards s origin .The emergence of this lesion on the tongue frequently happens with children, up to 2 years old. In spite of appearing in different places, the Hairy Polyp is more often seen in the anterior area of the dorsum . For CarAmong possible imaging examination methods, there are ultrasonographies, radiographies and magnetic resonance. The advantage of computed tomography is its fast acquisition time, which facilitates the sedation. In spite of that, the use of ionizing radiation is a factor that must always be kept to a minimum necessary in these patients. The use of iodated oral radiocontrastant, in a reduced amount, dripped on the tongue’s surface, aggregated the advantages of lessening the time of sedation and exposing the area to viewing with a much higher resolution. In addition, it permitted the prompt comprehension of the extension and mobility of the lesion, which used to vary between distal and medial positions, constantly adapting itself to the oral functions.The subsequently possibility of multiplanar and tridimensional reconstruction adds great academic value to the acknowledgment and comprehension of this pathology and its anatomical associations. In this case, it’s concluded that the whole outline of the process allowed the cylindrical mass, when positioned on the anterior part of the tongue, took on the functions of a pacifier. At a slight swallowing provoked by the contrast used, the lesion made a motion towards the opposite direction. Such a dislodgment, activated upon swallowing, creates favorable conditions for breathing and ingestion difficulties.It’s possible that suction, when the lesion is moved anteriorly, might have sped up the growth of the lesion, justifying the complaints and the urgency in the intervention. With its body getting more and more voluminous, it was clear that there was a constant mechanical obstruction of the oropharynx.The stimuli reflexes of coughing and liquid regurgitation, added to difficulties of the reflex between swallowing and breathing, hardened the patient’s feeding and led her into an increased irritability, expressed through crying. The presence of vomiting couldn’t be confirmed. The apprehension of this aspect at the location of the lesion equally guided the choices of orotracheal intubation and general anesthetic.This pathology deserves special attention since its existence, by obstructing the upper airways, compromises vital functions such as swallowing and breathing. Due to clinical similarities to other kinds of lesions, its recognition at the oral routine examinations of the child may sign to systemic diseases. The referred case brings out the Hairy Polyp as a process susceptible to fast development, making vital functions vulnerable, mainly with newborns. Therefore, it’s essential that the clinical diagnosis be accurate, so that the surgical ablative local treatment can be done, and the final anatomopathologic can be substantiated. The use of computed tomography with an oral radiocontrastant may not only aid in diagnostics, but also clarify the dynamics of the tumor.The postoperative follow-up is key, not only to check that the healing happens without recurrence, but also to watch the functional response of the tongue in a developing and growing patient.Written informed consent was obtained from the patient for publication of this report and any accompanying images.The authors declare that they have no competing interests.EP: surgery performed and documented the case. Drafted the manuscript. MBB: performed the histopathological analysis. BH: exams performed the analysis of the image. DP: performed surgery. HVA: performed the surgery and review of literature. Drafted the manuscript. MAM: conducted a review of literature. Drafted the manuscript. JHS: anesthesia carried out in obtaining the CT scan and at surgery. All authors read and approved the final manuscript.