We report two cases of unilateral upper eyelid swelling with multiple small lumps as an unusual complication of autogenous fat injection for cosmetic forehead augmentation.Two female patients were referred to our clinic for unusual unilateral eyelid swelling, with multiple small lumps. The duration of symptoms differed in each case, but both patients had a history of autogenous fat injection for cosmetic forehead augmentation at a local plastic surgery clinic. The lumps were small (diameter 5 mm~10 mm), palpable, hard, and nonmobile, and were evaluated by magnetic resonance imaging (MRI). Lumps from the eyelids of two patients were excised under general anesthesia. All of the masses were located deeply and found near the superior orbital rim or lateral orbital rim. The lumps exhibited chronic inflammation with fibrosis. Some of the lumps showed foamy histiocytic aggregation and foreign body lipogranuloma, resulting from iatrogenic fat injection. After excision, all masses and swelling disappeared, and moderate ptotic eyelid or lagophthalmos of affected eyes also improved.To our knowledge, eyelid swelling with multiple lumps in the eyelid is a very rare complication of autogenous fat injection for cosmetic forehead augmentation. This report should be helpful for ophthalmic clinicians who encounter these unusual symptoms. Autogenous fat is an easily accessible, renewable resource that can be harvested from multiple sites with little or no morbidity. After transfer to other locations, autogenous fat serves as non-allergenic, well-tolerated, supple, and versatile implant material. Therefore, autogenous fat injection into the periorbital or mid-face region is a common type of cosmetic surgery for rejuvenation in middle-aged and older Western subjects. In Asians, autogenous fat injection has become increasingly popular for forehead augmentation in relatively young people to enhance the esthetics of the typical flat Asian forehead. Although autogenous fat has good long-term safety and no severe adverse reactions, we encountered a case series of eyelid swelling with multiple mass-like lesions after dermal injection of autogenous fat. These complications are reported in order to assist clinicians in future diagnoses of patients after autogenous fat injection.A 26-year-old woman complained of eyelid swelling and blepharoptosis of the right upper lid for 1 month that had developed slowly over the previous 6 months. On examination, a small mass 2~3 mm in diameter was seen laterally and another small mass measuring 5~7 mm in diameter was seen medially; these masses were not easily movable and poorly demarcated. A medially placed mass was palpated deeply near the superior orbital rim. No tenderness or skin discoloration was noted. Moderate blepharoptosis of the right upper lid was observed (Figure A 30-year-old female visited our clinic for frequent upper eyelid swelling and a palpable medially located mass with lid lag and 2 mm lagophthalmos (Figure The clinical characteristics of two patients were summarized Table .Autogenous fat is an excellent material for filling spot defects, such as depressed scars, and area defects, such as trough and sulcus deformities. Autogenous fat is easily accessible for harvesting and is available in adequate quantities for injection, even in lean patients. The fat is readily accepted at the implantation site due to its autogenous nature. Facial Complications of autogenous fat injection include a visible lump, the collection of fat at the injection site, an uneven skin surface, and allergic reactions to albumin or anesthetic agents. A rare, but potentially devastating, complication is embolization with loss of vision or stroke, which has been recently reported. The visual loss can result from retinal ischemia caused by occlusion of the ophthalmic and central arteries by fat emboli,6. To ouFrequent eyelid swelling and multiple lumps with or without blepharoptosis are rare but potentially serious complications resulting from autogenous fat injection of the forehead. Here, we present the first report describing such adverse reactions and their treatment. Although blepharoptosis was not severe, if it progresses and does not improve with systemic anti-inflammatory medications, surgical intervention might be necessary, as it was in Case 1. Two patients were no longer suffered from frequent eyelid swelling after the masses were completely removed. Finally, if non-absorbable fatty tissue remains at the forehead, at the first injection site, it could move inferiorly and cause upper eyelid complications.Written informed consent was obtained from both patients for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor in Chief of this journal.The authors declare that they have no competing interests.JSP, WKC, KSP and SWY treated the patient and in doing so acquired the case data; they were also involved with drafting of the manuscript. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2415/13/32/prepub


Twenty-five to 45 percent of all schwannomas occur in the head and neck. Most of them arise along the vestibular portion of the eighth cranial nerve (acoustic neurinoma). They rarely originate from the peripheral facial nerve or other nerves within the parotid gland. Less than 4% of schwannomas involve the nasal cavity and paranasal sinuses. They arise from the branches of the trigeminal nerve and autonomic nervous system.We report two cases of schwannomas arisng from intraparotid facial nerve and nasal cavity. The first case diagnosed in a 62-year-old-man presented with 2-year-history of painless mass of the parotid gland. The lesion was found to be cystic through the pre operative examinations and investigations. The histology of the specimen retained an intraparotid cystic schwannoma. The second case concerned a 75-year-old-man presented with episodes of nasal obstruction, rhinorrhea, anosmia and headache. Histological study of the specimen showed fusiform cells with strongly and diffusely immunostaining for S100 protein suggesting a schwannoma.http://www.diagnosticpathology.diagnomx.eu/vs/1098335216112242. Schwannomas of the facial nerve can occur at any point along its complicated anatomical course from the cerebellopontine angle to its multiple peripheral branches in the face. They originate rarely from the peripheral facial nerve or other nerves within the parotid gland and they are typically present as an asymptomatic parotid mass . SchwannA 62-year-old man, presented with 2 years history of a painless and gradually enlarging right parotid cystic mass. He denied any facial weakness, twitching or pain. There was no history of prior irradiation or trauma or smoking. Examination revealed a 5 × 4 cm cystic non-tender mobile mass. Facial nerve function as well as the remainder of head and neck examination was normal. The neck ultrasonography showed a hypoechoic and homogeneous right intraparotidien process, though limited, measuring 6 cm of diameter. Dental panoramic showed osteolysis of the right mandible and computed tomography confirmed the origin of the intra-parotid lesion Figure .Total conservative parotidectomy was done. The branches of the facial nerve were identified. Intra-operatively, a cystic mass with solid component was found involving the parotid gland. The surgical specimen of 2.8 × 1, 8 × 0, 8 cm and 4, 5 × 4, 5 × 2 cm with a cystic appearance was examined. Histopathology showed spindle-shaped cells arranged in characteristic Verocay bodies Figure  and negaA 75-year-old man with episodes of nasal obstruction, rhinorrhea, epistaxis, anosmia and headache. CT scan showed a large polypoid mass in the maxillary sinus and prolapsed into ipsilateral nasal cavity. Magnetic resonance imaging (MRI) scan revealed an enhancing, cystic mass in the right nasal cavity measuring 7 × 5 × 3 cms hypointense on signal intensity area on T1-weighted images, and high intensity area on T2-weighted images. The tumor expands into the orbit and nasopharynx. Hitological study of the specimen revealed a composed of cellular Antoni A areas with Verocay bodies and hypocellular myxoid Antoni B areas. The cells are fusiform with elongated fribillary cytoplasm, and buckled to spindled nuclei which show little pleomorphism, There are frequently small to medium-sized vessels with hyalinization in the Antoni B areas Figure . The tumThe head and neck regions accounts for 25 to 45 percent of benign schwannomas, and most of these arise in the eighth nerve but are relatively uncommon from the seventh or the fifth nerves. Neurogenic tumors found are mainly neurofibromas and schwannomas (Neurilemmomas). They represent a pathology, which is often not taken into account during clinical practice .th) or from any other peripheral nerve running within the gland and it presents clinically as a primary salivary gland tumor [th) nerve and autonomic nervous system, and most commonly involve the ethmoid and maxillary sinuses, followed by the nasal cavity, sphenoid and frontal sinuses. Cellular schwannoma tends to be located in the midline [Schwannoma of the parotid gland can arise either from the facial nerve  or von Recklinghausen disease of the skin which is associated with somatic mutations at the NF1 gene, a tumor suppressor gene located in the pericentromeric region of chromosome 17 .Grossly, schwannomas are solitary, encapsulated tumors usually attached to or surrounded by nerve. It appears to push axons aside and degenerative changes like cystic alterations or hemorrhagic necrosis are usually present .The critical step in the management of neurogenic tumors of head and neck is the diagnosis. They may be also discovered incidentally on imaging studies.The use of fine-needle aspiration (FNA) to aid in the diagnosis is a common and appropriate practice in parotid schwannoma ,12. The Hence, a negative FNA finding alone should not delay or hinder surgical intervention when it is otherwise clinically indicated.Magnetic resonance imaging (MRI) with gadolinium is the study of choice for imaging the suspected nerve lesions of the parotid and nasal cavity. On T1-weighted images, the main trunk of the nerve appears hypointense, with hyperintense surrounding fat tissue, it appears on high intensity within T2-weighted images .Macroscopically, sinonasal and intraparotid schwannomas range in size up to 7 cm. they are a well-delineated but non-encapsulated globular, firm to rubbery yellow-tan mass. The cut surfaces show tan-grey, yellowish, solid to myxoid and cystic tissue, commonly with haemorrhage .Histologically, two microscopic patterns of schwannomas exist, Antoni A and Antoni B. Antoni A lesions are characterized by broad interlacing ribbons of extended spindle cells with elongated nuclei arranged in waves, drifts and whorls. On cross section, these cylindrical cells produce a palisading pattern of nuclei about a central mass of cytoplasm called a Verocay body. Antoni B pattern is made up of very loose tissue, lacking the arrangement in the bundle and palisades, and is thought to be a degenerative form of Type A with a looser texture and polymorphism of cells separated by abundant myxoid, often microcystic matrix .Mitoses are usually absent and malignant transformation of schwannoma is exceptionally rare . ImmunosThe differential diagnoses of intraparotid schwannoma should comprise the large diagnostic spectrum in which spindle cells are involved such as neurofibrome, fibroblastic/myofibroblastic tumors, most frequently nodular fasciitis, and fibromatosis with an infrequent myofibromatosis, fibroma, haemangiopericytoma, solitary fibrous tumor or inflammatory pseudotumor (inflammatory myofibroblastic tumor) .Intranodal palisaded myofibroblastoma is another tumor that must be taken into account in differential diagnosis .Like a cystic mass of the parotid, the most differential diagnoses include retention cysts, post-traumatic sialoceles, Warthin’s tumor, mucoepidermoid carcinoma and necrotic metastases . The difBenign facial and trigeminal nerves schwannomas grow slowly and resection is not always indicated. Several authors have reported the occurrence of facial paralysis or several nasal symptoms when a simple biopsy or careful resection with apparent preservation of the function nerve is indicated ,6.Schwannoma should be considered in the diagnosis of slowly growth lesions involving the parotid gland and nasal cavity.Written informed consent was obtained from each patient for publication of this report and any accompanying images.FNA: Fine-needle aspiration; MRI: Magnetic resonance imaging; SMA: Smooth muscle actin.The authors declare that they have no competing interests.IK, CL, HN and EH participated in the histopathological evaluation, performed the literature review, acquired photomicrographs and drafted the manuscript. HT and AA conceived and designed the study, gave the final histopathological diagnosis and revised the manuscript for important intellectual content. All authors read and approved the final manuscript.


The involvement of the ovary in lymphomatous process is rare. Such an involvement may occur in 2 ways, primary or secondary. We report 5 cases of ovarian non-Hodgkin's lymphoma, with 3 of which primarily arising in the ovaries. Ovarian lymphoma can mimic more frequently occurring tumors including advanced epithelial carcinoma and radical surgery may be performed instead of a biopsy. The immunophenotypic and clinicopathologic features exhibited in this small series are described to call attention to early diagnosis and treatment of ovarian lymphoma. All patients were diagnosed as having DLBCL after ovary biopsy. Different treatment modalities were used and prognosis of the patients was reported. Involvement of the ovary by malignant lymphoma, particularly Non-Hodgkin's lymphoma (NHL), is a well-known manifestation of disseminated lymphoma with a frequency of 7% to 26% . HoweverμL (the reference level: 142 × 10³ μL). Serum CA 125 and lactate dehydrogenase (LDH) were raised , but CA 19-9 and CEA were within normal limits. Transabdominal ultrasonography demonstrated a complex adnexal mass, measuring 12.5 × 11 cm with solid areas and internal echoes. A computer tomography (CT) scan revealed an 11 × 9 × 7 cm, solid, left ovarian mass with ascites  non-Hodgkin's lymphoma (diffuse large B-cell lymphoma) was made. Bone marrow biopsy showed no evidence of malignant cells. The patient was treated with 8 cycles of rituximab, cyclophosphamide, hydroxydaunorubicin, and Oncovin (R-CHOP) chemotherapy. Followup to the 20th month after the surgery showed no evidence of recurrence. The patient died from pneumonia at the 20th postoperative month.A 53-year-old grand multiparous woman presented with complaints of frequent urination, night sweats, progressing abdominal swelling, and increasing pain in the abdomen of 4-month duration. She had no significant medical or family history and she had used no medications. Physical examination revealed a tender mass of 20-week size in the lower abdomen. Laboratory studies showed elevated levels of CA125 and LDH, 648 U/mL and 454 IU/L, respectively. Other laboratory tests, including CA 19-9 and CEA, were within normal limits. Ultrasonography showed a large amount of free fluid in the cul-de-sac, a left adnexal solid mass measuring 10 × 6 cm, and a right adnexal solid mass measuring 6 × 5 cm, and those were confirmed on a CT scan. Exploratory laparotomy revealed a 10 cm right-sided and 8 cm left-sided solid masses and 15 cm retroperitoneal mass localized in front of the aorta and below the kidneys. At the time of the surgery, 3 L of ascites was evacuated. There were multiple implants ranging from 1 cm to 5 cm on the surface of peritoneum. Frozen section of the resected masses demonstrated a poorly differentiated malignancy. A total hysterectomy and bilateral salpingooopherectomy with omentectomy, lymph node sampling, appendectomy, peritoneal cytology, and peritoneal biopsy were performed. Immunohistochemical evaluation showed strong straining for CD20. These same cells were negative for pan-cytokeratin, S-100, EMA, CD30, CD10, CD5, HMB-45, and inhibin. Bone marrow biopsy showed no evidence of malignant cells. The diagnosis of diffuse large B-cell lymphoma was made and stage was assessed as IV BE according to the Ann Arbor system. After operation, MRI scans showed para-aortic and paracaval bulky lymph nodes. A chemotherapy protocol comprising 6 cycles of R-CHOP regimen was administered to the patient. Radiation therapy was also given for retroperitoneal mass. Followup to 6 months after surgery showed no evidence of recurrence.A 52-year-old, grand multiparous woman presented to our clinic with symptoms of fatigue and progressive abdominal swelling for 6 months. She had no significant medical or family history. A tender mass of 20-week size from the pelvis to the umbilicus was found on physical examination. CT scan revealed a large 16 × 12 × 10 cm solid right ovarian mass extending to the retroperitoneum with ascites. Serum CA125 was elevated (41 U/mL). Other laboratory tests including CA19-9, CEA, and LDH were within normal limits. On minilaparotomy, wedge of the mass was taken for histopathology examination. On immunohistochemical evaluation, CD20 showed strong staining, while CD5, CD3, and bcl-2 were negative. Histopathology of the ovarian tumour was reported to be consistent with diffuse large B-cell lymphoma. Bone marrow biopsy showed no evidence of malignant cells. The patient was assessed as stage I BE according to the Ann Arbor system. A chemotherapy protocol comprising 8 cycles of R-CHOP regimen was administered to the patient. Followup to 12 months after chemotherapy showed no evidence of recurrence.A 57-year-old grand multiparous woman presented to our clinic with a history of abdominal swelling, night sweats, and fatigue of 2 months. She had no significant medical or family history. Upon examination, a tender mass of 24-week size with restricted mobility was detected. CT scan revealed a 16 × 15 × 10 cm left ovarian mass with ascites . LaboratOvarian involvement by malignant lymphoma is well recognized and is seen at autopsy with a frequency of 7%–26% . HoweverLymphomas of the ovary can occur at any age, but most often in women over the age of 40  . Our patIt is difficult to determine whether ovarian lymphomas are primary or secondary. There has been much controversy in the histogenesis of POL. It is demonstrated that normal ovaries contain lymphocytes of B-cell and T-cell lineage within cortical granulomas and rare lymphocytes are present throughout the ovarian stroma and within ovarian follicles and corpora lutea, which can be the source of NHL . Fox et In conclusion, prognosis of ovarian lymphomas is often poorer than nodal lymphomas because of delayed or inaccurate diagnosis. The best treatment option seems to be chemotherapy. Physicians should be aware of this rare presentation to avoid radical surgery, which is unnecessary.


We present a case of pneumatosis intestinalis (PI) of the colon in the setting of inflammatory bowel disease that was treated with medical management rather than emergent surgery. While the reflex response to extraluminal air in the abdomen is abdominal exploration, consideration of the clinical context in which PI is discovered and an understanding of a complete differential diagnosis of the sources of PI is critical to avoiding unnecessary surgery. The small intestine 42%) is most commonly involved followed by colon 36%), with involvement of both in 22% . PI is a% is most6%, with A 60-year-old man was admitted for a flare of Crohn's disease, with pancolitis confirmed by colonoscopy with biopsy. He was discharged on oral prednisone, but was readmitted one week later for persistent abdominal pain, diarrhea, and a low-grade fever (38.1°C). A computed tomography (CT) scan of the abdomen on readmission showed thickening of the transverse, descending, and sigmoid colon. The right colon was normal at the time. After starting high-dose intravenous steroids (hydrocortisone 100 mg every 8 hours) and intravenous antibiotics (Cefazolin and Metronidazole), the patient defervesced and his symptoms improved. Two days later, a repeat CT performed for an elevated white blood cell count  revealed extensive PI of the right colon . BecauseIn PI, extraluminal gas predominantly localizes to the submucosal and subserosal planes of the small or large intestine, but can also localize to the muscularis propria . While tMost cases of PI are asymptomatic  such thaOverall, PI is a rare radiological finding and one that occurs in wide spectrum of clinical disorders. In the setting of the acute abdomen with coexisting systemic sepsis, necrotic bowel must be suspected and emergent operative management pursued. However, in the setting of a nonacute abdomen and a stable patient, benign causes of PI need to be considered in the differential diagnosis. As illustrated by the presented case, the radiological finding of PI is not always an indication for surgery and may be treated with medical therapy alone in many clinical circumstances.


Introduction. Rhabdomyomas of the head and neck are exceptionally rare benign mesenchymal tumors. Rare cases have been reported to involve other sites of the body including the head and neck regions. Case Presentation. We report a case of voluminous extracardiac adult rhabdomyoma affecting adult patients and initially seen as slowly growing, indolent neoplasms. The patient is a seventy-year old male Italian patient. Conclusion. Adult extracardiac rhabdomyoma is a rare benign tumor that may present with symptoms that vary from aerodigestive tract obstruction to remaining asymptomatic for many years. Although histology is very characteristic, several differential diagnoses have to be considered. To our knowledge, this is the first case of voluminous adult-type symptomless rhabdomyoma. Rhabdomyomas are benign mesenchymal tumours with skeletal muscle differentiation. They can be divided into extracardiac-and intracardiac-type depending on the localization. The extracardiac type is extremely rare, with a propensity for occurrence in the head and the neck and for multiple localization ; recurrences may be observed after complete excision (10 to 40%). They are virtually never associated with other malformations; on the other hand intracardiac rhabdomyomas are often associated with tuberous sclerosis  or other genetical disorders.Extracardiac rhabdomyomas can be divided into four clinically and morphologically different types: the adult-(AR), the fetal-(FR), the genital-type (GR), and rhabdomyomatous mesenchymal hamartoma. AR mainly affects elder people, with a mean age of occurrence of 60 years, with a 3 : 1 male : female ratio. Malignant transformation has not been described . FR is rA seventy-year old male patient with hypertension and asthma comorbidity came to our attention with a 8-9 cm mass in the left submandibular region. He had a history of alcohol abuse, and he had a smoking history of two to three packs of cigarettes per day for approximately 60 years. This mass appeared one year before, slowly growing, giving no pain to the patient. A cervical ultrasound was performed and described an oval hypoechoic mass which did not infiltrate the thyroid or the submandibular gland. Even a CT scan was performed. An aspiration biopsy from the tumor showed a smear of low cellularity dominated by large polygonal to round cells with abundant acidophilic granular cytoplasm. Chest radiographs showed no acute pulmonary process. The patient underwent a surgical excision of the mass via an open transcervical approach. During the procedure, the jugular vein seemed compressed and shifted forward by the mass which was in contact with the hyoid bone and with the upper pole of the left thyroid but did not infiltrate these structures. A 7 mm jp drainage was applied at the end of the procedure. The tumor was then sent to pathology: macroscopically it was a brown and brick red lobulated mass, was soft in consistency, and measured 8.5 × 2.5 × 1.5 cm . ImmunohExtracardiac rhabdomyomas are very rare tumors comprising less than 2% of the neoplasm of the striated origin. This entity was first described in 1897 by Pendl, who presented a case of fetal type , and fewAdult extracardiac rhabdomyoma is a rare benign tumor that may present with symptoms that vary from aerodigestive tract obstruction to remaining asymptomatic for many years until incidentally being diagnosed in surgery or post-mortem. It is most commonly found in the head and neck regions and should be considered in the differential diagnosis of masses in this region.


Colonic atresias are the rare malformations of the colon and constitute about 1.7 to 15% of all gastrointestinal (GI) atresias. A 6-month old infant presented with recurrent episodes of sub-acute intestinal obstruction since birth. During the index admission, patient had clinical signs of complete intestinal obstruction. The patient was operated and type I sigmoid-colon atresia found which on further exploration tuned out to be of perforated mucosal web variety. The resection of the involved part of colon and a primary end to oblique colo-colic anastomosis was performed.  There are three anatomical types of colonic atresia, the less frequent being type I. This is characterized by a mucosal diaphragm, completely occupying the lumen, without sero-muscular interruption . At timeA 6-month-old male infant presented to emergency room with abdominal distension, bilious vomiting and constipation for two days. There was a history of repeated episodes of such events since birth. On clinical examination the patient was febrile, with temperature of 100F, pulse 120/min and respiratory rate 28/min. Abdomen was distended with visible bowel loops. Bowel sounds were absent. Digital rectal examination revealed empty rectum. Abdominal radiograph showed air fluid levels  . In our Source of Support: NilConflict of Interest: None declared


Nasolabial cysts are nonodontogenic cysts that occur beneath the ala nasi. Its pathogenesis is uncertain. Because the nasolabial cyst is a soft tissue lesion, plain radiographs are useless. CT and MRI should be evaluated. In this report, a nasolabial cyst is described including its features on ultrasonography (USG) and CT exams. Nasolabial cysts are rare nonodontogenic cysts located adjacent to the alveolar process above the apices of incisors  and firsUSG is a diagnostic method that is often used for examination of soft tissue lesions and it can also be used in the maxillofacial region. Nasolabial cyst is a lesion of the soft tissue and it can be diagnosed with USG. In this case, CT and USG diagnosis have shown the presence of a 1.5 cm diameter cystic lesion in the soft tissue which has been diagnosed as nasolabial cyst in the histopathologic examination.A 38-year-old woman presented with a 5-year history of an increasing left nasal ala area swelling. She mentioned difficulty in breathing for about 3 months. She was suffering from pain in nasolabial fold for about 1 year. There was no history of trauma and no medical history. Clinical examination revealed a smooth and fluctuant mass in the left nasal ala area. A floating tumefaction in the nasolabial sulcus was observed. Intraoral examination revealed bulging of the buccoalveolar sulcus by the swelling. There was a facial asymmetry due to bulge on the left side of the nose. The associated teeth tested vital with electrical vitality testing. Nasolabial cysts generally do not cause bone destruction, but in this case bone destruction was observed on panoramic radiograph  and CT iKlestadt was the first person who reported the original theory of nasolabial cyst. According to his theory, the cyst is derived from epithelium trapped in the line of fusion between the lateral nasal and maxillary processes . Now theDifferential diagnoses of nasolabial cysts should be made with dentoalveolar cysts and incisive foramen cyst. They should also be differentiated from epidermoid or dermoid cysts, unilocular lymphatic malformations, and froncule and dental abscesses . VitalitNasolabial cysts are localised in the soft tissue and they usually are unobserved in conventional radiographies. However, in some cases, if bony destruction is involved, they can be observed radiographically. CT and MRI can be used for diagnosis, localization, and defining the borders of nasolabial cyst. CT is more frequently used in comparison to MRI due to lower costs .USG is another diagnostic method in maxillofacial surgery. USG is used for diagnosis of cellulitis and abscess in the maxillofacial region , 10. AlsUSG is inadequate to reflect intrabony defects and lesions and generally used for examination of the soft tissues. It can be safely used in the maxillofacial region to visualize soft tissue lesions like nasolabial cyst. In this case, USG examination of the patient who applied to our clinic displayed a cystic lesion in the soft tissue. Having nonionized character and being inexpensive and more available are some advantages of USG over MRI and CT. In addition to this, the experience of the doctor who performs the USG is an important factor in defining the borders of the lesion accurately. In this case we have shown that USG is useful in the diagnosis of nasolabial cyst. Finally we conclude that direct graphy and USG are adequate in cases in which nasolabial cyst is suspected.


Granulosa cell tumors (GCTs) of the ovary account for 2 to 5 of ovarian malignancies. We present two patients with malignant ovarian adult GCT. In one patient, a combination of bleomycin, etoposide, and cisplatin was effective after initial surgery for malignant GCT. In the other, an aromatase inhibitor was effective for recurrent malignant GCT. We also review the literature for further management of this tumor. Because GCT of the ovary is rare, it will be necessary to elucidate the clinical phenotype and establish treatment protocols by accumulating and analyzing more patients. Granulosa cell tumors (GCTs) of the ovary comprise 2 to 5% of malignant ovarian tumors ,2, and 2A 26-year-old woman presented to her doctor in December 2010 with abdominal pain and bloating. She had never been pregnant, but had a history of surgery for patent ductus arteriosus (at the age of 1 year) and also had type 2 diabetes. A pelvic neoplasm was suspected, and the patient was referred our department in January 2011 for detailed investigation and treatment.Abdominal ultrasonography showed a mass, with hyperechogenicity. On magnetic resonance imaging (MRI) scan, an irregular, solid mass, 130 mm in size, was seen in the peritoneal cavity with associated pelvic ascites. Computed tomography (CT) showed enlargement of the left subclavian, para-aortic, and pelvic lymph nodes, and metastasis was thus suspected. No abnormality was seen with peripheral blood tests and biochemical tests, but the level of a tumor marker, CA125, was raised .A diagnosis of malignant ovarian tumor was made, and surgery was performed. The laparoscopic findings indicated that the mass was an irregular, solid tumor derived from the left ovary. Because multiple distant metastases were present, a left adnexectomy was performed.The cut surface of the tumor was yellowish and friable Figure . On path2 on days 2, 9, and 16; etoposide 100 mg/m2 on days 1 to 3; cisplatin 15 mg/m2 on days 1 to 5) every 3 weeks.Based on these results, a malignant GCT of the ovary  stage IV) was diagnosed. After surgery, the patient was given three cycles of chemotherapy , with one cycle every 3 weeks, which resulted in a reduction in the size of the lesion. However, re-expansion of the deposits occurred, and total resection of the intraperitoneal recurrences was attempted in October 2006  on day 1) every 3 weeks.An abdominal CT performed in February 2004 showed an intraperitoneal mass, indicating tumor recurrence. The patient was given 10 cycles of chemotherapy  1 mg/day, but a CT scan in September 2010 showed recurrent intraperitoneal masses. Because enlargement was also seen, a third, complete resection was attempted in May 2011. Pathological examination showed atypical cells proliferating in a mixed solid and cystic manner in all specimens in the recurrences obtained in 1998, 2006, 2009, and 2011, and Call-Exner bodies, coffee bean-like cells, and mitoses were also visible Figure . ImmunohBased on the above results, a malignant GCT was diagnosed. Oral administration of anastrozole 1 mg/day has continued with the patient followed up as an outpatient, and this has resulting in disease-free survival to the most recent follow-up (April 2012).et al. reported a patient with a very aggressive ovarian GCT, composed of granulosa, sarcomatoid, and fibrothecomatous tissues [Ovarian GCT is classified as a sex cord-stromal tumor and is treated as a borderline malignant tumor. These tumors show the clinical and pathological features of malignancy at a rate of 20 to 30%, and comprise 2 to 5% of ovarian malignant tumors ,2. Progn tissues . The tum tissues .The histopathological characteristics of GCTs include cells with a coffee bean-like longitudinal nuclear groove and a micro-follicular structure termed the ‘Call-Exner body’. In patients with malignancy, a large number of mitoses and dyskaryoses are also seen. According to one report, the number of mitoses is a pathological prognostic factor . Alpha IBecause it is difficult to distinguish GCTs from epithelial ovarian tumors pre-operatively, and because the accuracy of intraoperative rapid pathological diagnosis is not good, basic techniques used for the management of ovarian cancer  and staging laparotomy are recommended as the initial therapy . IndicatPatient 1 had a reduction in the size of lesions in lymph nodes after BEP therapy, confirming the efficacy of the treatment. However, because serious interstitial pneumonia also occurred in this patient, as has been reported for other cases in which bleomycin was implicated in treatment-related death, the use of this treatment needs to be reconsidered. By contrast, even though the intraperitoneal tumor recurred in patient 2 after the start of aromatase inhibitor administration, the tumors were small, suggesting that the aromatase inhibitor slowed tumor growth, and that this drug is effective against recurrent tumors.The results for our two patients suggest that BEP therapy is effective after initial surgery and that an aromatase inhibitor is effective for recurrent tumors. However, because GCTs are rare, it will be necessary to elucidate the clinical phenotype and establish treatment protocols by accumulating and analyzing more patients.Written informed consent was obtained from the patient or the patient’s family for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interest.RT, YY, TY, YM, KA, RT and HM treated the two patients as a team and reviewed the literature. RT drafted the manuscript, and YY, TY, YM, KA, RT and HM also helped to draft the manuscript. All authors read and approved the final manuscript.RT, TY, YM, KA and RT work as instructors at Department of Obstetrics and Gynecology, Hirosaki University Graduate School of Medicine. YY is an associate professor and HM is a professor and is also chair of the Department of Obstetrics and Gynecology, Hirosaki University Graduate School of Medicine.


The patient underwent laparotomy to remove the uterine fibromyoma. Intraoperatively, a voluminous soft mass arising from the Retzius space was found; it was firmly adhered to the uterus with obliteration of vesicouterine pouch owing to severe adhesion to the anterior surface of uterus. The tumour was isolated, enucleated from the prevesical space, and removed; moreover, the patient became asymptomatic after surgery. In conclusion, leiomyoma of the Retzius space is a very rare benign tumour that should be considered in the presence of severe bladder voiding symptoms.Extrauterine leiomyoma is a very rare clinical condition; we report a case of leiomyoma of the Retzius space in a 49-year-old women who suffered for two years from bladder voiding symptoms characterized by dysuria, feeling of incomplete emptying, and pelvic pain. Clinical evaluation and abdominal and transvaginal ultrasound suggested the presence of a voluminous (about 10 cm in diameter) fibromyoma of the anterior uterus surface. The urodynamic evaluation demonstrated the presence of bladder outlet obstruction (voiding pressure greater than 20 cm H Primitive tumours of the Retzius space are infrequently in women; although the neoplasms in most of the cases are characterized by benign histology, isolated cases of aggressive cancers of adjacent tissue  can involve the Retzius space , 2. The tumours may remain asymptomatic for a long time becoming very large when detected. Common symptoms are related to urinary voiding secondary to bladder compression; they include dysuria, frequency, or hesitance in urination, urinary retention. A rare case of a giant mass of the Retzius space diagnosed in a woman with urinary voiding symptoms is herein reported.2O and maximum flow rate less than 12 mL/s) with a postvoiding urine residual equal to 80 mL; moreover, the presence of cystocele and urethral stricture was ruled out by performing clinical evaluation, cystography and cystourethroscopy. A 49-year old woman presented with a clinical history dated from two years of bladder voiding symptoms characterized by dysuria, feeling of incomplete emptying, and pelvic pain. On physical examination the vaginal examination, was painless and uterus was fixed with an irregular surface due to a voluminous anterior mass. The abdominal and transvaginal US showed normal ovaries and a mass of 10 cm in diameter localized in the anterior zone of the uterus; the ultrasound pattern suggested the presence of a uterine fibromyoma that compressed the bladder wall. The urodynamic evaluation demonstrated the presence of bladder outlet obstruction  , 4 and aleiomyoma of the Retzius space constitutes a rare cause of urinary voiding difficulties due to obstruction of the urethra or/and bladder , and uptPreoperatively, clinical evaluation and US, as first choice, in most of the cases are not able to distinguish tumours of the Retzius space from the most common fibromyoma of the uterus; therefore, the indication to perform a more accurate radiological imaging (MRI or CT scan) for the suspicion of a pelvic mass often remains unheeded and only intraoperatively the right diagnosis is performed.In conclusion, leiomyoma of the Retzius space is a very rare benign tumour that should be considered in the presence of severe bladder voiding symptoms.


The tumor was circumscribed but unencapsulated and showed obvious hemorrhagic necrosis. Microscopically, the tumor was composed of patternless sheets of alternating hypercellular and hypocellular areas of spindle cells displaying mild to moderate nuclear atypia, frequent mitoses up to 8 per 10 high power fields, and a 20% Ki-67 proliferative index. Immunohistochemical studies revealed reactivity for CD34, CD99 and vimentin, with no staining for all other markers, confirming the diagnosis of SFT. No areas of dedifferentiation were seen after extensive sampling. Based on the pathologic and immunohistochemical features, a diagnosis of de novo malignant renal SFT was warranted. Our report expands the spectrum of malignant progression in renal SFTs. Even though this patient has been disease-free for 30 months, long-term follow-up is still mandatory.The kidney is a relatively infrequent site for solitary fibrous tumor (SFT). Among the previously reported cases, only two cases of malignant renal SFT developing via dedifferentiation from a pre-existing benign SFT have been reported. Here we reported a case of  AlthougA 50-year-old woman was admitted to our hospital with one-month history of soreness and pain in her right flank, without gross hematuria or other constitutional symptoms. Laboratory findings were unremarkable. Physical examination revealed a palpable right flank mass. A computed topography (CT) of the abdomen showed a huge necrotic tumor occupying the perirenal space of right kidney without evidence of either local invasion or lymphadenopathy Figure . The patA nephrectomy specimen  with attached ureter and perirenal fibroadipose tissue was received. The specimen was bisected to reveal a 9 × 9 × 6 cm circumscribed but unencapsulated tumor occupying the perirenal space of the upper and middle poles of kidney. The tumor was firm and showed a yellowish white to tan-gray, myxoid and lobulated cut surface with prominent hemorrhage and necrosis Figure . Microscde novo malignant solitary fibrous tumor was established.Immunohistochemically, the tumor showed weak CD34 positivity Figure  and diffSFT is a relatively uncommon but distinctive mesenchymal neoplasm, originally described in the pleura cavity and later reported to occur ubiquitously . Althougde novo occurrence or (2) dedifferentiation or sarcomatous overgrowth from a pre-exsisting histologically benign SFT [de novo since we did not find any areas of dedifferentiation after extensive tumor sampling.Malignant behaviors in the form of recurrence and/or metastases can occur in 10% to 15% of intrathoracic SFTs and up to 10% of extrathoracic SFTs ,3. Malignign SFT . Most renign SFT . To our nign SFT ,8. A detThe criteria for clinical malignancy in intrathoracic SFT, first proposed by England et al. in 1989, include increased cellularity, pleomorphism, mitotic count more than 4 per 10 high power fields, necrosis, hemorrhage, size more than 10 cm, non-pedunculated and atypical locations  . The diaSFTs show a wide variety of microscopic growth patterns and should be distinguished from benign and malignant spindle cell tumors. Positive immunoreactivity for CD34 and CD99 is characteristic of SFT, and highly valuable in differentiating from other mesenchymal tumors ,4. HowevThe prognosis of extrapleural SFTs is more unpredictable than that of pleural tumors due to lack of large-scale studies. The clinical outcomes were rather strikingly different in the two malignant renal SFTs with dedifferentiation. While a patient developed multiple small lung nodules 4 months post-operatively , the othWritten informed consent was obtained from the patient for publication of this case report and accompanying images. Submission of this case report was approved by Institutional Review Board (IRB) of Chang Gung Memorial Hospital, Tao-Yuan, Taiwan. A copy of the written consent is available for review by the Editor-in-Chief of this journal.SFT: solitary fibrous tumorThe authors declare that they have no competing interests.TYH was responsible for data collection, literature search and manuscript preparation. YCCC carried out gross and microscopic examinations. WHC performed the surgery and clinical follow-up of the patient. CSC interpreted pre-operative and follow-up imaging studies. CLC, CCH and HPC participated in the microscopic analyses and helped making a final diagnosis. JRC participated in manuscript preparation and approved the final manuscript. All authors read and approved the final manuscript.


Dear Sir, Most of the intussusceptions in infants are idiopathic in nature. The incidence of intussusception decreases as the age advances. On the contrary, the frequency of lead point (secondary cause) increases with age. Meckel's diverticulum is the commonest anomaly of small bowel. There is 5% incidence of complications in patients having Meckel's diverticulum. Inverted Meckel's diverticulum, as a lead point for intussusception, is a rare entity and commonly reported in adults. Pediatric cases are scarcely reported in literature [1-4]. A 2-year-old girl presented with one day history of abdominal distension, bilious vomiting and not passing stool. There was no preceding history of significant illness. The patient was vitally stable. Abdomen was distended with tenderness on right side. Digital rectal examination revealed an empty rectum. Abdominal radiograph showed air fluid levels. Ultrasound of abdomen gave an impression of intussusception. Laboratory investigations were within normal limits. Patient was managed with intravenous fluids, prophylactic antibiotics, nasogastric decompression, and analgesics. Exploratory laparotomy was performed within 3 hours of presentation that revealed an ileoileal intussusception which was manually reduced. A polyp like pedunculated growth was palpable at about 30 cm proximal to ileocecal valve. An enterotomy was performed for polypectomy; however, the pedunculated growth had a pale tissue at the tip giving a suspicion of heterotopic tissue . The involved segment of the ileum was resected and end to end hand sewn anastomosis was performed. The polypoid growth was inspected carefully and with a suspicion of inverted Meckel's diverticulum, reduced back confirming the diagnosis . The patient had an uneventful recovery. She remained well at one year follow-up (1 year). Histopathology of the specimen revealed ectopic pancreatic tissue in the inverted Meckel's diverticulum.Pantongrag-Brown et al [2] found that 21% of Meckel's diverticulae were inverted, of which 72% presented with small bowel intussusception; however, overall incidence of intussusceptions due to Meckel's diverticulum is about 4%. The exact mechanism of inversion of Meckel's diverticulum is not known but it is believed to result from hyperperistalsis generated due to irritation of enteric nervous system by activity of the ectopic tissues which is found in more than 50% cases of Meckel's diverticulum. In more than 85% of cases with heterotopy, gastric mucosa is present; pancreatic tissue is rarely reported in inverted Meckel's diverticulum as a lead point for intussusception. Meckel's scan, radio labeled RBC scan, superior mesenteric angiography, and CT scan are important diagnostic tools. On CT scan, a mass located in the distal small bowel, having a collar of soft tissue attenuation around a central area of fat attenuation, gives strong suspicion of inverted Meckel's diverticulum [1-4].I believe that inversion of Meckel's Diverticulum could be a normal phenomenon in persons having it. It could revert, possibly, in the majority without being symptomatic. In few cases, inversion could advance to the extent which results in intussusception. Rarely, it could fix inside the bowel due to mucosal inflammation and swelling which may preclude the reversion; such inversion presents as bleeding per rectum (ectopic tissue), pain abdomen, intestinal obstruction secondary to intussusception (commonly) or mass (in case of hypertrophy) [1-5]. This theory plausibly explains the various presentations of inverted Meckel's diverticulum however further studies are required for scientific evaluation of this hypothesis. In cases where inversion advances to intussusception, Meckel's diverticulum could be reduced back along with the rest of invaginated bowel. In the index case, inverted Meckel's diverticulum could not be reduced with the invaginated bowel indicating a fixed inversion of Meckel's diverticulum. It simulated a pedunculated polyp, however after excision, the suspicion of ectopic tissue and careful dissection of the specimen made us identify it.Source of Support: NilConflict of Interest: None declared


Retrorectal tumors are a rare group of neoplasms that occur most commonly in the neonatal and infant population. They vary in presentation, but teratomas are the most common and often present as a protruding mass from the sacrococcygeal region. Immediate surgical resection is indicated when found and coccygectomy is performed to prevent recurrence. When teratomas recur, the patients most often have vague symptoms and the tumors usually have malignant transformation. Here, we present the case of a young woman who underwent surgical resection of a sacrococcygeal teratoma at 3 days of age where the coccyx was not removed. She presented at 31 years of age with lower extremity paresthesias and radiography revealed a cystic mass extending from the sacrum. After resection, pathology revealed a recurrent teratoma with nests of adenocarcinoma. Retrorectal neoplasms refer to a rare and heterogeneous group of tumors that occur in what is known anatomically as the retrorectal space. This is a potential space defined superiorly by the rectal peritoneal reflection, anteriorly by the rectum, posteriorly by the sacrum, and inferiorly by the rectosacral fascia. Although the retrorectal space does not technically include the more inferiorly located supralevator and deep postanal spaces, lesions occurring within these areas are still generally referred to as retrorectal or presacral neoplasms.As mentioned, retrorectal neoplasms are rare and occur more commonly in the neonatal population with a female : male ratio of 3-4 : 1 . They haOf the congenital lesions, teratomas usually are found in the sacrococcygeal region and they continue to be the most common tumor found in newborns where 80% are found in the first six months of life , 7. TheyA 31-year-old obese female presented with a 6-month history of intermittent pelvic pain along with occasional right lower extremity paresthesias. Her only significant medical history was that at birth she was found to have a congenital sacrococcygeal teratoma . She undThe mass was approached operatively with the patient in prone position and through a parasacrococcygeal incision. It was found to be a well-encapsulated cystic lesion and it was completely resected en block with the coccyx. Histologic sections demonstrated an invasive mucinous adenocarcinoma with pools of mucin and moderately to well-differentiated malignant glands coming to within 1 mm of the resected margin. The tumor cells showed immunoreactivity to CDX2, a marker of intestinal differentiation, and coexpression of keratins CK20 and CK7. Smaller areas of teratoma were also identified and consisted of a pancreatic parenchyma, salivary gland tissue, and gut wall. The gut wall was composed of intestinal type mucosa with an underlying layer of smooth muscle resembling muscularis mucosa. These findings are consistent with a teratoma demonstrating endodermal and mesodermal components . BecauseTumors that occur in the retrorectal space are challenging because they present with vague symptoms; they are often inaccessible on physical exam, and the anatomic region is not well known to most physicians. Because this “potential space” contains residual totipotent stem cells, a wide variety of neoplasia can develop here and it can present throughout life. There are 2 clinical patterns most commonly observed: (1) neonates presenting with large, predominantly benign tumors that are mature or immature teratomas and (2) infants and children <4 years presenting with teratomas containing malignant cells . In the When sacrococcygeal teratomas are found in the neonatal patient, physical examination and radiologic assessment are used to guide resection. The surgeon must try and avoid nerve injury as bowel and bladder incontinence can occur if damage occurs to the bilateral sacral nerve roots proximal to S3 , 11. ConThese tumors are rare and there is a lack of consensus data for postoperative chemotherapy and radiation. It has been shown that, with early detection and surgical excision, these tumors can be treated with excision and coccygectomy only . If therThis case is unique because it demonstrates that not only an infant born with a SCT can be seemingly cured with surgery in the neonatal period but also that recurrence can happen decades later. The recurrence can manifest in the anatomic region known as the retrorectal space and the presenting symptoms may be vague. This case also supports the prior findings of increased rates of malignancy in recurrent lesions. Finally, it reinforces the notion that a history of prior resection for SCT in the neonatal period should be noted by physicians caring for the patient even when the index operation was done long ago.


Odontoameloblastoma (OA) is an extremely rare mixed odontogenic tumor with both epithelial and mesenchymal components. OA develops from proliferating odontogenic epithelium and mesenchymal tissue. Till date, around 20 cases have fulfilled the histologic criteria of the current World Health Organization (WHO) histologic classification of odontogenic tumors. It affects predominantly young patients with a median age of 20.12 years, and has a predilection for males and occurs in posterior segments of either jaw with slight inclination for mandible. Review of literature shows only three reported cases in the anterior mandible. Here, we report a case of OA in the anterior mandible. Odontoameloblastoma (OA) is an extremely rare neoplasm, which is defined by World Health Organization (WHO) and Philipsen & Reichart as follows: “A neoplasm that includes odontogenic ectomesenchyme in addition to odontogenic epithelium that resembles an ameloblastoma (SMA) in both structure and behavior. Because of the presence of odontogenic ectomesenchyme, inductive changes take place leading to the formation of dentin and enamel in parts of the tumor”.et al., 1983), calcified mixed odontogenic tumor , soft and calcified odontoma , and ameloblastic odontoma .[Several names exist for this kind of tumor in the literature, which include odontoblastoma , adamant-odontoma  and OA.OA affects predominantly young patients with a mean age of 20.12 years in the reported cases, appearing up to 59% in patients under 15 years of age. There is a slight male predilection. This tumor usually occurs in the posterior segment of either jaw, with a slight inclination for mandible.4 Only thThe tumor exhibits growth characteristics similar to ameloblastoma and presents centrally destructive lesions. Symptoms include a slow progressive swelling of the alveolar plates, dull pain, an altered occlusion, delayed eruption or impacted teeth.464Radiological examination usually reveals a multilocular radiolucency with radiopaque areas resembling mature dental tissue. It commonly exhibits a well-defined margin, displacing the surrounding erupted teeth rather than producing root resorption.Treatment wise, OA should be treated by wide excision and closely followed up for at least 5 years, as there2The exact incidence of this neoplasm is difficult to determine as the current information on this unusual lesion comes from isolated and sporadic case reports. In many The aim of this article is to present a rare case of OA in the anterior mandible.A 47-year-old male patient reported to the Department of Oral and Maxillofacial Pathology, with the chief complaint of a rapidly growing swelling on the back side of the anterior lower jaw since 3 months. The patient gave the history of similar lesion at the same site which recurred twice in the last 7 years, for which he underwent surgical procedures. No record was available with the patient of the previous treatments.Intraoral examination revealed a mucosal colored swelling of approximately 3 cm in diameter on the lingual aspect of mandible on the right side, extending from 32 to 44. The swelling was firm, non-tender, non-fluctuant, non-compressible Figures  and 2. IA provisional diagnosis of calcifying epithelial odontogenic tumor was considered. Routine hematological investigations were within normal limits. The tumor was carefully excised and the specimen was sent for histopathologic examination.Histopathology showed the following: odontogenic epithelial cells arranged in the form of follicles and stellate reticulum like cells in the center which are surrounded by ectomesenchymal cells (4×) ; tall coOA is an aggressive odontogenic tumor. The lesion has generated much confusion and controversy in the literature as evidenced by the reporting of developing odontomas, AFOs and OA under the terms ameloblastic odontomas, admantoodontona and soft and calcified odontomas. To clarify the confusion, WHO deleted ameloblastic odontoma from its “histologic typing of odontogenic tumors, jaw cysts and allied lesions”, and subdivided this category into AFOs and odontoameloblastoma. This separation was based on the differences in the clinical and histological behavior of the lesions.Review of literature of 22 cases of OA showed the histologic criteria of unequivocal ameloblastoma, mature connective tissue, fragments of malformed calcified dental structures .OA is a very rare mixed odontogenic neoplasm characterized by the simultaneous occurrence of an ameloblastoma and a compound or complex odontoma in the same tumor mass. The epithelial proliferation forms islands or intermingled cords that produce the follicular or the plexiform patterns typical of ameloblastoma, but unlike conventional ameloblastoma, these induce the production of mineralized dental tissues on the adjacent mesenchymal cells and may respond to these changes with the production of enamel.This tumor is not exclusive of the human race and has also been described in sheep, monkeys, cats, and rats. Some OAsEven though very few cases have been reported, it is suggested that OA possesses clinical and microscopic features that allow differentiating it from typical ameloblastomas and odontomas. First, it tends to occur with equal frequency in the mandible and maxilla, while ameloblastoma is prevalent in the mandible. Secondly, OA tends to produce bone expansion similar to ameloblastoma, while odontoma seldom produces swelling of the affected region. Some OAs present intermittent or dull pain which is not usually referred in other benign odontogenic tumors. A case of peripheral OA has been reported by Palaskar in the region of 37, 38.[Histologically, it is difficult to differentiate between OA, AFO and ameloblastic fibrodentinoma (AFD). AFO and


A 18-year-old male presented with low back ache. The patient was investigated and was diagnosed to have painful scoliosis. X-ray and other examinations could not reveal any diagnosis. The patient was referred to undergo bone scan on clinical suspicion of osteoid osteoma and to rule out stress fracture if any. Planar bone scan was performed, which showed a lesion in L3 vertebra and was further evaluated with SPECT (Single photon emission computed tomography) study to characterize the lesion. On SPECT examination, the classical features of osteoid osteoma, the double density sign (11), was noted in the pars interarticularis region. These findings were confirmed by a CT scan, which showed a sclerotic lesion in pars interarticularis of L3 vertebra. The patient was posted for operation and was relieved of symptoms in the postoperative follow-up. Osteoid osteoma is one of the most common benign tumors of the spine and it is also the most common cause of painful scoliosis in adolescents.–3 Up to Two thirds of spinal osteoid osteomas represent as painful scoliosis. It is, however, very common to miss the diagnosis and the patient may be subjected to treatments, such as bracing, adding discomfort to the patient.An 18-year-old male presented with low back ache since 1 year. The patient had scoliosis. Typical evening raising plain X-ray was done and was inconclusive. Bone scan was ordered to evaluate boney pain.A three-phase bone scan was performed. The planar images revealed a focal solitary lesion in the L3 vertebra. The lesion was further evaluated with SPECT study for further characterization, which revealed a lesion in the L3 vertebral pars interarticularis region. Central focal hot spot with peripheral decremental uptake was noted in L3 vertebral pars interarticularis.A CT scan was performed to precisely localize the lesion and to confirm the diagnosis. CT scan showed osteosclerotic lesion in the L3 vertebral pars interarticularis region Figure –4.Osteoid osteoma is a benign osteoblastic neoplasm most often seen in young males, frequently in the first 3 decades of life and may be found in the cortical or cancellous bone with a further 5% reported as subperiosteal, and multicentric foci were also reported.5Preliminary diagnosis carries some difficulties, mostly due to unawareness of the condition, leading to delay in the management. The first imaging procedure is usually the plain radiography,6–8 which4671Although radioisotope imaging is not pathognomonic for osteoid osteoma, it can be very useful in diagnosis and anatomic location, and intraoperative and postoperative identification.–57–10 Th55A CT scan is essential for optimal planning before surgery is performed and for postoperative confirmation of complete resection.Treatment can be surgical or conservative. To achieve a surgical cure the entire nidus must be removed completely; medical management of osteoid osteoma is well recognized and is usually needed whenever the lesion is inaccessible even with the most carefully planned surgical exposures, or is adjacent to vital neurovascular structures, or when the patient refuses any operative interference.In conclusion, any child or adolescent with scoliosis complaining of back pain, particularly long-standing, should be investigated thoroughly, keeping in mind the possibility of osteoid osteoma and a bone scan should be ordered as a primary investigation because a plain radiograph may not be significant in detecting these lesions.


The purpose of this report is to present the findings in a case of squamous cell carcinoma (SCC) of the conjunctiva which was the initial sign of systemic cancers. A 94-year-old woman without known systemic diseases developed a mass in her right conjunctiva. She was referred to our hospital 5 months after the onset. She was diagnosed with conjunctival SCC by biopsy. Systemic CT before the surgery revealed multiple liver lesions, lung legions, and a large mass surrounding the appendix. The patient requested the surgery, and the main aim of the surgery was cosmesis. Histopathological examinations of the specimen led us to the final diagnosis as SCC. She did not receive any other therapy because of her age. As no other surgical procedures were undertaken, it is uncertain as to whether the conjunctival lesion was primary or secondary. Although, it is extremely rare that SCC of the conjunctiva is the initial sign of systemic cancers, careful systemic examinations to find other cancers should be made. A squamous cell carcinoma (SCC) of the conjunctiva accompanied with systemic cancers is very rare . Only onA 94-year-old woman had noticed that the conjunctiva of the right eye was hyperemic 10 months earlier. She was suspected of having a conjunctival tumor at a private clinic 5 months later but she refused to undergo further examinations because of her age. After a rapid increase in the size of the mass, she was referred to the Chiba University Hospital. At the first examination, her visual acuities were hand motion at 30 cm OD and 0.8 OS. Slit-lamp examination showed a large, irregular-surface mass on the nasal conjunctiva. The mass was so large that it covered the pupil of the eye Figures . The ocu Because the size of the tumor was large, we performed a systemic CT examination. The CT examination showed a large tumor surrounding the appendix, multiple legions in the liver, and lungs . No othe Because the patient requested surgery for cosmesis, we excised the tumor and used 0.04% Mitomycin C eyedrops 2 times/day. The tumor was almost completely excised , and theCervantes et al. reviewed 287 cases of SCC of the conjunctiva, and only two cases had regional metastasis (0.7%) . Grossni The patient did not undergo chemotherapy because of her age, and the purpose of the operation was cosmetic. However, the patient and her family were very satisfied with the outcome of our surgery. Thus, even when the life span cannot be extended by a surgery, the quality of life can be significantly improved. Thus, although the patient died one month after the operation, we believe that our decision of performing tumor excision in this case was appropriate and meaningful for the patient and her family. It is extremely rare that a conjunctival tumor is the initial manifestation of systemic cancers . More co


Odontogenic fibromyxoma is a rare and locally-invasive benign neoplasm found exclusively in the jaws. It has the potential for extensive bony destruction and extension into the surrounding structures.In the presented case, radiographic and histological features as well as the treatment and follow-up of odontogenic fibromyxoma accompanying odontogenic cyst of mandible in an 8-year-old boy are discussed. Almost 75% of odontogenic myxomas occur in patients around 23-30 years of age with a slight female predilection. It rarely occurs in patients over 50 or under 10 years of age.4 The tumor occurs almost equally in the maxilla and mandible with a slight predilection for the posterior mandible.1 A few cases are described in the ramus, condyle and non-tooth bearing areas.Odontogenic myxoma is a rare and locally-invasive benign neoplasm found exclusively in the jaws. It is a benign odontogenic tumor and should not be confused with soft tissue myxoma, which is a relatively common tumor of the soft tissues. Odontogenic myxoma usually occurs in the tooth-bearing areas. Therefore, it is supposed to be of tooth origin, and derived from the mesenchymal portion of a tooth germ—most likely of the dental papilla.5 The more common fibrotic odontogenic myxomas  have larger tumor bodies, which makes their curettage easier.Radiographically, the majority of cases present as expansile mass and multilocular radiolucency with or without scalloped borders, although some are unilocular, and rare cases present with a diffuse and mottled appearance which can be mistaken for a malignant neoplasm.7Histologically, odontogenic myxoma is made up of loose and delicate fibrous connective tissue. The fibroblasts are stellate and are suspended on a delicate network of collagen fibrils.Although odontogenic cysts are relatively common in the jaws, co-occurrence of odontogenic myxoma with an odontogenic cyst is a rare finding, as reported in the present case.An 8-year-old boy was referred to a private clinic of oral and maxillofacial surgery. The patient suffered from a mild swelling (mass) with tenderness on the right side of mandible for a period of six month. The skin over the swelling area was normal and there was no local rise of temperature. On palpation, the mass was immobile with bony hard consistency. Buccal and palatal cortexes were expanded and there was no history of paresthesia or hyposthesia. The lesion extended from mandibular right second premolar to the half height of the mandible ramus.The panoramic radiograph revealed a well-defined, unilacullar radiolucency involving mandibular right second molar and a multilacullar radiolucency on the distal of mandibular right first premolar to the mesial of the right second molar.The treatment consisted of surgical excision of the lesion under local anesthesia with frozen sections during the surgery sent for pathological examination to reveal whether the lesion is benign or malignant and the margins are free of lesion or not. After several frozen sections, clear bony margins were reached to prevent recurrence of the neoplasm. Mandibular right second molar was also extracted. Pathologic lesion involved a neurovascular bundle  that was separated meticulously from the lesion.3 .The biopsy also contained the crown of a molar tooth. Fibromyxomatic tissues consisting of bundle and round cells with blood vessels and collagen fibers in a bright matrix were seen. In some areas of the lesion, structures of odontogenic cyst with inflammatory features were found(The biopsy consisted of an amorphous, irregular mass sized 3.5 × 2 × 0.2 cmre found.Immediate post-operative follow-up was scheduled weekly for one month, monthly for the next five months and twice a year for the next three years. In the first week, anesthesia or hypoesthesia was not detected. A panoramic radiograph 4 weeks after surgery showed relative healing of the surgical site. Panoram8 The radiographic features are variable, and the diagnosis is, therefore, not easy.8 According to variable clinical and radiographic appearance, it should be considered in the differential diagnosis of radiolucent lesions of both jaws in all age groups.11 According to the radiographic feature, site, and consistency of lesion in the present case, the differential diagnosis was odontogenic myxoma, mural ameloblastoma and infected odontogenic cyst.12 However, according to the pathological findings of the case, final diagnosis was odontgenic fibromyxoma accompanied with odontogenic cyst.Odontogenic myxoma is a rare and locally invasive benign neoplasm found exclusively in the jaws. It commonly occurs in the second and third decades of life and the mandible is involved more commonly than the maxilla. The lesion often grows without symptoms and presents as a painless swelling.Being rare in children, occurrence of odontogenic fibromyxoma in the first decade and its combination with odontogenic cyst was the most considerable finding of this case.


Background. Conventional osteochondromas are common bone lesions developing in the metaphyseal region of growing skeleton. Marginal excision is the treatment of choice for such tumours. Extraosseous cartilaginous tumours are rare and their biological potential is poorly characterized. Case Presentation. A-52-year old woman presented with 3-year history of fullness and dull pain and inability to flex her left knee, sit cross-legged, or squat. Clinical and imaging studies revealed a nodular mineralised mass in the anterior portion of the knee displacing the patellar tendon laterally. Excision biopsy confirmed the diagnosis of extraosseous osteochondroma-like soft tissue mass. There is no recurrence at two-year followup. Conclusion. An integrated clinicopathological diagnosis helps to clarify the nature of extraosseous cartilaginous tumour that can arise at an unusual anatomic site. Complete surgical excision is the treatment of choice. Conventional osteochondromas are common bone lesions developing in relation to the periosteum and occur around the growth plate of long bones, especially the knee . They teSynovial chondromatosis is a less-common proliferation of multiple nodules of hyaline cartilage within the synovium, in most cases requiring thorough synovectomy .Para-articular or extraosseous osteochondromas are rare in older individuals  and theiHere we report one more case of extraosseous osteochondroma-like soft tissue mass in the anterior portion of the knee joint.A-52-year old woman presented with 3-year history of fullness and dull pain involving the left knee. She had been aware of a mass in the knee joint that had been progressively increasing in size. She was unable to flex her knee, sit cross-legged, or squat. There was no history of trauma or constitutional symptoms.On physical examination, there were fullness on the medial aspect of left knee and palpable nodular mass of approximately 6 × 4 cm over the infrapatellar region medial to the patellar tendon . The masUnder regional anaesthesia with the patient supine in a bloodless field furnished by a pneumatic tourniquet, a 7 cm longitudinal incision was made medial to the patellar tendon. The mass was lying medially and behind the patellar tendon in Hoffa's fat pushing the tendon laterally  and impiPostoperative recovery was uneventful. Sutures were removed on the 12th postoperative period. The patient returned to her daily activities regaining full flexion within two weeks after surgery. She was on regular followup. When last reviewed 24 months after excision, she was asymptomatic with no clinical and radiographic signs of recurrence of the lesion (Figures Grossly, the resected specimen measured 7 × 6 × 5 cm with a large cartilaginous cap and was surrounded by adipose tissue and a fibrous capsule. Histologically, the cut sections showed numerous lamellar bony trabeculae with mature cartilaginous cap. Fibrofatty tissue was seen at the periphery . There wThere are several different types of lesions composed of bone and cartilage that occur around joints. A conventional osteochondroma arises from a developmental defect in the growth plate, resulting in an osteocartilaginous proliferation in which the bony stalk, continuous with that of the bone of origin, typically grows away from the nearest joint , 16. MosExtraosseous osteochondromatous lesions are rare. Usually arising from the joint capsule or para-articular soft tissue without attachment to bone, these lesions may be large with clinical and radiological features of malignant process. These have been reported earlier as para-articular osteochondroma, intracapsular chondroma, intra-articular osteochondroma, extraosseous osteochondroma, soft tissue osteochondroma, capsular osteoma, ossification of infrapatellar fat pad, and ossifying chondroma –14.The concept of para-articular osteochondroma was first introduced in 1958 by Jaffe , who useOnly few intra-articular osteochondromas have involved the anterior and more rarely the posterior knee joint space , 9, 14. At MRI, there were no irregularities or thickening of the cartilaginous cap greater than 1 cm. Hence, there was no suggestion of malignant feature . The borIntraoperatively, the tumour was completely intra-articular with no evidence of continuity to bone. The gross appearance and histological examination demonstrated the features of an extraosseous osteochondroma-like soft tissue mass with secondary bone formation similar to normal enchondral growth . The benMRI is recommended for further characterization of nature and extent of an intra-articular osteochondroma. Operative removal is the procedure of choice when function is reduced and nature of tumour is uncertain.In conclusion, an integrated clinicopathological diagnosis helps to clarify the nature of extraosseous cartilaginous tumour that can arise at unusual anatomic site. Complete local surgical excision is the treatment of choice.


Kidney lesions may be difficult to diagnose only by radiological exams, often requiring proof by tissue biopsy. Moreover, if enlarged regional lymph nodes are also present, the spectrum of differential diagnoses is even greater. The role of regional lymph node dissection in this setting is not clearly established. We show the case of a patient with a kidney mass associated with a conglomerate of para-aortic and iliac lymphadenopathies corresponding to an oncocytoma and a nodular lymphocyte predominant Hodgkin' lymphoma, respectively. Diagnosis of these two lesions was performed by morphology and immunohistochemistry. This case reflects how imaging can mislead to diagnosis and how histological confirmation helps decide treatment management. Renal oncocytomas are uncommon benign neoplasms of the urinary tract, accounting for 3% to 9% of all renal tumors . Most caA 76-year-old woman with no significant past medical history was admitted because of a new rapid atrial fibrillation and congestive heart failure due to an underlying coronary heart disease. A computed tomography (CT) was requested to rule out pulmonary embolism. Incidentally, a large left kidney mass was discovered. The CT showed the 9 cm diameter lesion localized in the midregion of the left kidney, with central areas of low attenuation and a conglomerate of left para-aortic and iliac lymphadenopathies . No otheThe patient did not report any pain, weight loss, B symptoms, or haematuria. On physical examination, bibasal rales and bipedal pitting edema were noted. No red blood cells were found in the urinalysis. The hemoglobin, serum creatinine, and urea were also normal.A malignant kidney tumor was suspected. The patient was addressed at our oncology department to discuss treatment. Because of the bulky lymphadenopathies, the urological surgeon did inquire about the role of neoadjuvant treatment. Considering the lack of histological diagnosis and the underlying heart disease, no indication for targeted treatment was retained, and a surgical resection was proposed. A total left nephrectomy with para-aortic lymphadenectomy by median laparotomy was performed. The anatomopathological report revealed a 8 × 8 × 7 cm tan-brown nonencapsulated tumor in the left kidney with central fibrosis. Histologically, well-differentiated neoplastic cells were seen with abundant eosinophilic granular cytoplasm, compatible with oncocytoma. The multiple para-aortic lymph nodes showed a nodular growth pattern and a background of lymphohistiocytes with a phenotype B CD20+. Complementary immunohistochemistry exams confirmed a nodular lymphocyte predominant Hodgkin' lymphoma (NLPHL). The staging evaluation concluded to a stage IIA for the NLPHL, and the oncocytoma showed no distant metastasis. Only clinical and radiologic followup was proposed.In our case, the initial radiological interpretation of the renal oncocytoma was challenging. Morphological characteristics were typical for renal cell carcinoma (RCC) as well, leading to prioritize a surgical resection for what was thought to be a malignant lesion. As retroperitoneal lymph nodes were voluminously present, a sampling lymphadenectomy was also performed. The anatomopathological report revealed two unexpected diagnoses.There is no clear data in the literature concerning benefit of lymph node dissection in RCC. It is well known that the survival of patients with regional lymph node metastasis is similar to that of patients with distant metastatic lesions . Thus, iThis case illustrates how alone imaging can be misleading. A biopsy could have been performed, but that would not have avoided the nephrectomy, as the kidney lesion was voluminous. Lymph node resection permitted to diagnose a lymphoma instead of a metastatic state of the presumably RCC, changing completely the treatment approach. Lymph node dissection was indicated in this situation, because it revealed crucial diagnostic information. No oncologic treatment should be delivered based on assumptions without histological confirmation. Assessments in a multidisciplinary meeting including a medical oncologist, a urologist, and a radiologist can help to avoid regrettable pitfalls.


Myxofibrosarcoma is a myxoid variant of malignant fibrous histiocytoma that most commonly involves the extremities of elderly people. However, a primary myxofibrosarcoma with bone invasion in young adults is extremely rare. Herein, we report the case of a 31-year-old male with a gradually enlarging left thigh mass, who had a history of left femur fracture and received an open reduction and internal fixation with titanium alloy plates and screws 33 months previously. Imaging investigations revealed an irregularly shaped soft tissue mass around the left femur shaft and a partial bone defect in the middle one-third of the left femur. Pathological examination of the resected specimen showed a multi-nodular appearance, abundant myxoid matrix and elongated curvilinear capillaries. Immunohistochemical studies revealed that the tumor cells was positive for VIM and MDM2, and was negative for CK, MSA, SMA, DES, S-100 and CD34. Labeling index of Ki-67 was 25%. Based on the morphological finding and immunostaining, it was diagnosed as a low-grade myxofibrosarcoma. The clinical and imaging examinations did not reveal the evidence of a primary cancer elsewhere, and the patient had no personal or family history of malignancy. To our knowledge, this is the first case of a primary myxofibrosarcoma developed following a fracture and metal implantation in young adults.http://www.diagnosticpathology.diagnomx.eu/vs/1745984882113605The virtual slide(s) for this article can be found here:  Myxofibrosarcoma is a myxoid variant of malignant fibrous histiocytoma (MFH), characterized by a nodular appearance, prominent myxoid matrix, elongated curvilinear capillaries, and location mostly in dermal and subcutaneous tissues in the extremities of elderly people-3. In 19Several clinical studies of myxofibrosarcomas showed that approximately 80% of the tumors occurred in the extremities and about 12% in the trunk, with a high incidence in the dermal or subcutaneous tissues. Other sHerein, we report a low-grade myxofibrosarcoma with left femur invasion in a 31-year-old male, and present the clinical, radiological and histopathological characteristics of this tumor. The clinical and imaging examinations did not reveal the evidence of a primary cancer elsewhere, and the patient had no personal or family history of malignancy. It is noteworthy that the patient had a history of left femur fracture and received an open reduction and internal fixation with titanium alloy plates and screws 33 months previously. To our knowledge, this is the first report on a primary myxofibrosarcoma that developed following a fracture and metal implantation in young adults.A 31-year-old male patient has a history of a multiple fracture involving the left femur shaft, the middle and distal segment of the left tibiofibula, as a result of a traffic accident in October 2008. Subsequently, the patient underwent an open reduction and internal fixation (ORIF), using titanium alloy plates and screws. Ten months after ORIF, an X-ray examination showed no displacement of the fracture site, and the bridging callus was visible on two standard views with partial obliteration of the fracture line. However, the patient did not undergo reoperation to remove the metal implants due to financial difficulties.In August 2011, the patient complained of a 3-month history of a gradually enlarging left thigh mass. Physical examination revealed a 12 × 10 cm firm, tender and fixed mass in the anteromedial left thigh, with local superficial venous engorgement and skin temperature increment. Percussion pain in the axial direction of the left lower limb was absent. The inguinal lymph nodes were not palpable. Routine chest radiograph and abdominal ultrasound examination showed no abnormalities. Other routine tests, including plasma biochemistry, liver function tests and urinalysis, were normal.The X-ray examination demonstrated an ill-defined osteolytic lesion with disappearance of the medial cortical bone in the middle of the left femur shaft, as well as an ambiguous image of a soft tissue mass. Computed tomography (CT) images clearly showed a partial bone defect approximately 9.7 cm in length on the medial border of the left femur shaft. A 9.7 × 11 × 19 cm irregularly shaped soft tissue mass was observed around the left femur, with the surrounding tissues compressed and shifted significantly  and negative for cytokeratin (CK), suggesting a mesenchymal histogenesis Figure a and b. The patient received three cycles of postoperative adjuvant chemotherapy with a combination of ifosfamide, doxorubicin, cisplatin and mesna, and he experienced no special discomfort during chemotherapy. In June 2012, the patient underwent the third operation for the removal of metal implants due to implant breakage in an accidental fall, followed by external fixation.However, in June 2013, only 22 months after tumor resection, a firm and painless mass approximately 5 cm in diameter was palpated in the medial left thigh. Magnetic resonance imaging (MRI) showed an irregular soft tissue mass with ill-defined margin in the anteromedial left thigh, accompanied by surrounding soft tissue swelling and normal muscle structure disappearance. Additionally, bone destruction in the left femur shaft was also observed Figure . Tissue Myxofibrosarcoma, also known as a myxoid variant of MFH, is one of the most common sarcomas in the extremities of elderly people, characterized by a high frequency of local recurrence. Distinctive histological features included the following: a commonly nodular growth pattern; a myxoid matrix containing elongated, curvilinear capillaries; and fusiform, round or stellate tumor cells with indistinct cell margins, slightly eosinophilic cytoplasm, and hyperchromatic atypical nuclei. These lThe etiology of myxofibrosarcoma remains unknown. It was previously reported that 2 patients developed a myxofibrosarcoma after radiotherapy,19. In tThere were some literatures about the tumors developed following a bone fracture. URIST reportedCase reports of malignant tumors at the site of metal implants in humans and domestic animals have implicated carcinogenesis as a potential, although rare, complication of metal orthopedic implants. The actual incidence of tumors in association with metal implants cannot be reliably estimated, considering that clinicians and veterinarians are not obliged to report adverse reactions to implants. Although the possibility that the apparent association is coincidental cannot be excluded, orthopedic surgeons should be aware of the potential carcinogenic risk of metal implants.In summary, we described a case of low-grade myxofibrosarcoma with bone invasion, which developed following a fracture and metal implantation in a young adult. To our knowledge, this case is the first report of primary myxofibrosarcoma in association with titanium alloy implants, which should alert doctors to the possible carcinogenic risk of titanium alloy implants.Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.CD34: Cluster of differentiation 34; CD68: Cluster of differentiation 68; CK: Cytokeratin; CT: Computed tomography; DES: Desmin; MDM2: Murine double minute 2; MFH: Malignant fibrous histiocytoma; MRI: Magnetic resonance imaging; MSA: Muscle-specific actin; ORIF: Open reduction and internal fixation; SMA: Smooth muscle actin; VIM: Vimentin.The authors declare that they have no competing interests.WSL, DL and QCY collected the data, planned and drafted the manuscript. WSL and XSZ were the pathologists who evaluated the specimen. SHL was the radiologist who provided the radiology images. CLH was the clinician who cared for the patient and provided the clinical data. All authors read and approved the final manuscript.


Chordomas are tumors derived from cells that are remnants of the notochord, particularly from its proximal and distal extremes, they are mainly midline and represent approximately 1% of all malignant bone tumors and 0.1 to 0.2% of intracranial neoplasms. Chordomas involving the sellar region are rare. Herein, we describe a 57-year-old male patient presenting with a history of retro-orbital headache, progressive loss of vision, and clinical features of hypopituitarism, for over 2 months. During evaluation, the CT scan revealed a large contrast-enhancing intrasellar tumor with a 3.6-cm largest diameter. The patient underwent transsphenoidal partial resection of the tumor, and histological examination was consistent with the diagnosis of chondroid chordoma. Although chordomas are rare, they may be considered to constitute a differential diagnostic of pituitary adenomas, especially if a calcified intrasellar tumor with bone erosion is diagnosed. Chordomas are midline slow-growing tumors that arise from proximal and distal extreme remnants of the notochord and represent 1% of all malignant bone tumors and 0.1 to 0.2% of intracranial neoplasms . They arThree established categories of intracranial chordomas were described based on anatomical location and clinical features: sellar chordomas associated with chiasm compression and hypopituitarism; parasellar tumors characterized by oculomotor nerve palsy, optic tract compression, and hypopituitarism; tumors involving the clival region and presenting with bilateral sixth cranial nerve paresis and brainstem compression . ChordomIn this paper, we report a patient presenting with a largely intrasellar chordoma simulating a nonfunctioning pituitary adenoma.A 57-year-old man reported retro-orbital headache, diplopia, progressive loss of vision (predominantly on the right side), fatigue, nausea, sexual dysfunction, dry skin, sleepiness, and constipation over a period of two months. He also had a history of systemic arterial hypertension and was using a pacemaker due to chagasic heart disease. In his familial background, he reported diabetes mellitus, thyroid disease, a death caused by stroke, and no cases of tumors.On physical examination, the patient presented postural hypotension and dry skin, any other alteration was not observed. Ophthalmological evaluation demonstrated a right-sided papillary defect, and visual field examination revealed a left temporal hemianopsia and a right/right-side blindness. μIU/mL, RV: 0.4–4.0; free T4 0.51 ng/dL, RV: 0.8–1.9; FSH 1.45 mIU/mL, RV: 0.7–11.1; LH 0.23 mIU/mL, RV: 0.8–7.6; total testosterone <20 ng/dL, RV: 181–758); cortisol and IGF1 were normal ; GH 0.62 ng/mL (RV: 0.0–5.0); beta-chorionic gonadotropin (hCG), alpha-fetoprotein (FP), and carcinoembryonic antigen (CEA) were normal; diabetes insipidus investigation also was normal. It was not possible to realize a dynamic evaluation about the diagnosis of GH deficiency. The computed tomography (CT) scan revealed a large heterogeneous contrast-enhanced intrasellar mass (largest diameter: 3.6 cm), with suprasellar extension, invasion of the sphenoid sinus, and erosion of the sellar floor : 2.5–17); central hypothyroidism, and hypogonadism . The serum sodium normalized after fluid restriction. Four months later, the patient evolved with intense headache and significant worsening of the left-side vision. A frontal craniotomy was performed for total removal of the tumor. However, an aneurism of the anterior circulation was ruptured during the surgery. Therefore, the aneurism was clipped, but the tumor could not be removed. He stayed 19 days in the intensive care unit to receive pneumonia treatment, and he died two months later.We report the case of a patient presenting with a chondroid chordoma that had an aggressive clinical course.Chordomas involving the sellar region are extremely rare. According to a recent review, only 22 patients have been described as possessing primarily intrasellar chordoma since 1960 and 43% of the patients were primarily diagnosed with a pituitary adenoma . This raThe skull base chordomas tend to present osseous permeation, and have a high rate of recurrence , 6. Thei The only known prognostic study published in 1993 estimated overall survival rates of 51% and 35% at 10 and 20 years, respectively . The imaging method of choice for the pituitary gland is MRI, but coronal CT with contrast and thin slices may be obtained in patients who have contraindications to MRI. On unenhanced CT scan pituitary adenomas are isodense to adjacent brain parenchyma and show intense contrast enhancement after injection of contrast material. Bony erosions are well demonstrated in bone window images. A sellar chordoma CT scan may reveal a contrast-enhancing intrasellar mass with bony erosions and calcifications . The immunohistologic profile of chordoma cells is reactivity to cytokeratins, vimentin, and epithelial membrane antigen (EMA) –33. ApprTranssphenoidal surgery , followed by radiotherapy, has been the accepted treatment for sellar chordomas , 17, 35.Although sellar chordomas are rare, they should be included in the differential diagnosis of tumors in the sellar region especially if a calcified tumor with bone erosion is revealed.


Our presumptive diagnosis was a massive mural thrombus and a concomitant chronic thromboembolism. Intravenous heparin and recombinant human tissue-type plasminogen activator was subsequently administered. However, the patient needed an emergency operation for sudden aggravation of the vital signs, and the tissue diagnosis was intimal sarcoma with poor clinical outcomes.Intimal sarcoma of the pulmonary artery is a rare malignant tumor that may be misdiagnosed as chronic pulmonary thromboembolism, even if various imaging techniques are used. We report a case of a 58-year-old man with pulmonary artery intimal sarcoma. Intimal sarcoma is a rare mesenchymal tumor, and it can arise from the great arteries. The incidence of pulmonary artery intimal sarcoma is almost twice that of the aorta . It usuaThis tumor is highly malignant and the prognosis is very poor. Survival is usually 12 to 18 months after the onset of the symptoms . The dia3, but the rest of the work-up revealed no hematologic evidence of intrinsic hypercoagulability. The N-terminal pro-B-type natriuretic peptide level was 395 pg/ml  and D-dimer was 0.816 μg/ml . The chest radiograph revealed atherosclerotic cardiovascular change and mild cardiomegaly. Electrocardiography showed a sinus rhythm.A 58-year-old man was admitted with a month’s history of exertional dyspnea, which had been worsening for twenty days. The patient was being medicated for diabetes mellitus and hypertension. He was relatively stable, at New York Heart Association functional class II-III. On physical examination, the patient exhibited tachypnea, with 20 breaths per minute and his oxygen saturation was 92%, his pulse was regular at 95 beats per minute, and his blood pressure was 130/80 mmHg. Auscultation revealed a prominent second heart sound and grade 3/6 systolic murmur over the pulmonary area. A blood count revealed platelets at 57,000/mmEvaluation with transthoracic echocardiography demonstrated a left ventricular ejection fraction of 56%. The right ventricle was enlarged and severely hypokinetic with a mobile echogenic mass in the supravalvular area of the main pulmonary artery. The color Doppler revealed a moderate tricuspid regurgitation. The systolic pulmonary arterial pressure was estimated with 70 mmHg. Subsequent evaluation with contrast-enhanced computed tomography (CT) of the chest demonstrated a large filling defect within the main pulmonary artery, which extended to near total occlusion of the left and right pulmonary artery, separately Figure . There h18F-fluorodeoxyglucose uptake in positron emission computed tomography (FDG-PET) was performed to confirm the neoplastic features and the distant metastatic extent of the disease. The results of the FDG-PET showed a large mass, but no definite uptake in the mass, and no other hypermetabolic lesions were noted elsewhere  due to the unstable vital status.Permanent histological findings revealed a non-distinctive myxoid spindle cell sarcoma with a rather lobulated, exophytic growth pattern. Some mitotic activity was seen and pleomorphic tumor cells surrounded the endothelium, in particular Figure . It was Given the poor prognosis and inability to wean the patient from the ECMO, we decided to withdraw the life support system after 5 days.The clinical features of pulmonary intimal sarcoma are similar, and it is frequently misdiagnosed as chronic pulmonary thromboembolism. It may include right heart failure due to pulmonary hypertension, along with such as a cough, dyspnea, chest pain, hemoptysis, and syncope. However, if concomitant symptoms include fever, cachexia, and anemia but no risk factors for venous thrombosis, malignancy should be considered. In our case, our initial diagnosis was pulmonary thromboembolism due to laboratory tests and radiologic features. There was no hematologic evidence of intrinsic hypercoagulability, but D-dimer was 0.816 ug/ml .Advances in imaging technology, such as echocardiography, contrast-enhanced computed tomography, and magnetic resonance imaging, have supported better diagnoses of masses in the pulmonary artery, but without always differentiating successfully between a thrombus and a malignancy. The FDG-PET is a more reliable method for the confirmation of the neoplastic features and the distant metastatic extent of the disease, due to increased FDG uptake in its malignancy. Even though, FDG is not a tumor-specific agent, FDG-PET is increasingly used. Several authors have reported that FDG-PET has been useful in a diagnostic workup for differentiating among or staging malignant diseases, and monitoring the response to treatment of pulmonary artery intimal sarcoma ,5. AmongIntimal sarcoma of the pulmonary artery is a rare malignant tumor that may be misdiagnosed as pulmonary thromboembolism, even if advanced imaging technology is used. Even though the FDG is poorly taken up in PET, the possibility of malignancy should be considered. We report a case of pulmonary artery intimal sarcoma that was not initially diagnosed as a malignancy due to poor uptake of FDG in a PET study.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.DL, DS and SP wrote the draft of the manuscript and obtained the written consent. TJ, JL and JC performed the literature review and participated in the manuscript writing and helped to the final writing of the paper and gave final approval of the manuscript. All authors have read and approved the final manuscript.