Papilloma virus infections of the oral cavity have been long recognized with various clinical expressions characterized as verruca vulgaris, Heck’s disease, multiple papilloma and condyloma acuminata. In this paper, we are highlighting a case of verruca vulgaris involving the oral cavity with extensive skin lesions in a nonimmunocompromised 9-year-old boy. Different treatment modalities are discussed in this article. Verruca vulgaris is a relatively uncommon oral lesion which is caused by an infectious agent HPV (human papilloma virus). Verruca vulgaris (VV) of oral mucosa is rare and is typically a childhood problem. The lesions of verruca vulgaris are circumscribed, firm, elevated papule with papillomatous hyperkeratotic surface. They occur most commonly on the dorsal aspect of the fingers and hand. They are also found on sole of feet. Lesions of verucca occur extensively in immunocompromised host. Here, we present a case of severe form of verruca in a healthy individual.A 9-year-old boy reported to the Department of Oral Medicine, Radiology and Diagnosis with multiple, small, whitish growth on his legs, hands, fingers and face . The firExamination of the oral cavity revealed whitish solitary lesion on right buccal mucosa with finger like projection and multiple small (4-5 in number) lesions were also appreciable on lower labial mucosa and on alveolar ridge in the region of lower left lateral incisor and canine . On palpThe patient underwent a complete blood investigation including Elisa test, which were within normal range. Excisional biopsy was performed with the patients parents consent (as he was a minor) from two different sites, i.e. dorsum of hand and intraorally from the right buccal mucosa. Histopathological examination revealed acanthosis with proliferative, hyperkeratotic, stratified squamous epithelium showing finger like projection and elongated rate pegs converging towards the center, producing cupping effect suggestive of verruca vulgaris. There were foci of vacuoled cells referred to as koilocytotic cells. Vertical tier of parakeratotic cells and foci of clumped keratohyalin granules were also seen . Patient1. Verruca vulgaris, condyloma acuminate, Heck’s disease, squamous papilloma are all local papillary lesions that share similar clinical feature yet their microscopic patterns differ.12 Verruca vulgaris commonly occurs in children and usually regresses spontaneously over a period of time.23 Papilloma virus type 2 and 4 are the most prevalent isolates from cutaneous warts, and these same viral genotypes are identifiable when verruca valgaris arise on the lip or on mucous membrane. Whereas condylomas are usually diagnosed in teenagers and young adults and are more frequently encountered in the mucosa membranes of the anogenital region, it may also be seen in the oral cavity perhaps arising therein as a consequence of oragenital sexual transmission.1 They are bigger in size and appear pink to red as a results of less keratinization.A variety of papillary lesions occur in the oral cavity and while many can be differentiated on the basis of histopathological features, they may closely resemble one another clinically4 There have been very few reported cases of verruca vulgaris in a nonimmuno-compromised patients. Thus, presented case is unique as there was no underlying immune supression.The case herein represent classical form of oral verruca vulgaris with extensive multiple skin involvement. Such type of exacerbated involvement is usually observed in immunocompromised host.45 Kirchner has speculated that a certain kind of lesion is more frequently found before puberty and links its presence to a genetic defect that retards the establishment of an effective defense mechanism until puberty leaving some children more susceptible than others to the infection.6 Verruca vulgaris may not require any treatment, but is done for cosmetic purposes and to prevent their spread.7From clinical observation, we found that the lesions were mainly located on the buccal mucosa, lower labial mucosa, also autoinoculation from a lesion located on hand has been observed. A slightly higher incidence was encountered for OVV in children and young adults and differs from other reports.8 The patient’s sibling’s lesions vanished in a similar way and in the present case also, no treatment was initiated as the patient denied any treatment.Most people develop an immune response that causes verruca vulgaris to vanish by themselves.78 Oral lesions are successfully surgically excised or they may be destroyed by laser, cryotheraphy or electrosurgery. Recurrence is seen in a small proportion of treated case. Verruca vulgaris does not transform into malignancy with or without treatment and two third will disappear spontaneously within 2 years especially in children.7 Detailed genetic study should be carried out to rule out any genetic origin in all the suspected cases.Skin verruca vulgaris is treated effectively by liquid nitrogen, cryotheraphy, conservative surgical excision or curettage or topical application of keratinolytic agents containing salicylic acid and lactic acid.in situ hybridization/ southern blot hybridization should be carried out. Fortunately in our case patient did not have any of this life threatening condition. The association of oral lesion with HPV should be extensively examined to clarify the pathogenesis of HPV infection in oral cavity.Due to paucity of cases of oral verruca vulgaris being reported, information regarding malignant potential is limited. In the current case despite an extensive skin lesion there was no evidence of underlying disease (AIDS/ dysplasia on histopathology). On basis of a single case, it is very difficult to infer that they may occur in nonimmuno-compromised patient and have no malignant potential. More studies need to be carried out to corroborate that they occur in immunocompromised patient. Any patient with presence of extensive verruca vulgaris lesion must be eyed with suspicion of AIDS/compromised immune system or internal malignancy and thorough blood investigation including ELISA/immunohistochemistry/


Primary cardiac tumors are uncommon and cardiac osteosarcoma is a rare disease. While complete surgical resection is considered to be the best treatment option for cardiac osteosarcomas, local and metastatic recurrences present challenges and indicate a poor prognosis. A combination of surgical resection with radio- and/or chemotherapy is a more effective course of treatment for osteosarcoma. In the present case, the patient underwent a complete resection of a primary cardiac osteosarcoma, and received chemotherapy and radiotherapy following local recurrence and metastasis to the bone post-operatively. Following these treatments, a rectal metastatic tumor was detected as causative of anemia. There is currently a lack of guidelines on the treatment of metastatic osteosarcomas in the intestine and there are few reports on rectal metastases. The present study described a laparoscopic resection of the osteosarcoma. The patient recovered without any complications and radiotherapy and chemotherapy were administered post-surgery to treat the bone metastases. The patient remained healthy at a follow-up examination, 61 months post surgery. To the A 41-year-old male was admitted to the Osaka Medical Center for Cancer and Cardiovascular Diseases  with paraplegia and anemia in March 2013. The medical history of the patient indicated that a surgical resection of a cardiac osteosarcoma had been performed at another hospital in September 2008. In May 2010, the patient presented with heart failure and recurrence of the sarcoma in both atria. Subsequently, the patient underwent a second surgical resection for the recurrent heart sarcoma, together with radiotherapy of the heart, and chemotherapy consisting of adriamycin (ADR), ifsofamide (IFO) and gemcitabine (GEM), post-operatively. Positron emission tomography-computed tomography (PET-CT) revealed multiple bone metastases in July 2011, and therefore radiotherapy to the mandible and scapula, Sr89, and Pazopanib were added to the treatment regimen. In December 2012, magnetic resonance imaging revealed metastases in the thoracic vertebrae, causing paraplegia to develop in March, 2013. Radiotherapy to the thoracic vertebrae Th4-9 and chemotherapy  were administered to treat the metastases, together with nerve decompression surgery to the thoracic spinal nerves of the Th4, Th9 vertebrae. Although the paralysis was controlled by these treatments, the patient was anemic, as indicated by a blood test. A fecal occult blood test (FOB-test) was performed to determine the cause of the anemia and the positive results predicted a rectal tumor. Colonoscopy revealed a rectosigmoid tumor as the cause of the bleeding, but this tumor did not have a pit-pattern typical of rectal cancer . ContrasHistological examination of the biopsy sample revealed growth of spindle cells with irregular nuclei. Immunohistochemical examination showed positive staining for Ki67 in 5–10% of the cells. These findings were compatible with a histological diagnosis of a metastatic osteosarcoma. To avoid bleeding from the tumor and future obstruction of the intestine, a laparoscopic rectectomy following indocyanine green marking  was perfThe patient recovered without any complications, and radiotherapy to thoracic vertebrae 12 to lumba vertebrae 5, and chemotherapy with ADR and cisplatin were administered post-operatively to treat the bone metastases. The patient was discharged from the hospital on postoperative day 67 and remained alive 61 months after the initial operation. The patient is still receiving chemotherapy and radiotherapy for the bone metastases.Life-threatening consequences of primary malignant cardiac tumors include obstruction to the intracardiac blood flow, interference to valve function, arrhythmias, and pericardial tamponade resulting from local invasion ,8,13–17.The patient in this case has been alive for 61 months following the first surgical resection, and 37 months after the presentation of multiple metastases. This is considered long-term survival. Upon careful analysis, this study proposes that repeating the surgical resection is the optimum choice of treatment as it is necessary to remove the rectal metastasis to prevent anemia and future obstruction To the best of our knowledge, this is the first reported case of using laparoscopic resection of a rectal metastasis. Laparoscopic surgery is minimally invasive and feasible for this type of metastatic tumor. In the present case, the patient recovered without any complications and was able to undergo chemotherapy and radiotherapy shortly after the repeated surgical resection. The literature on sarcoma metastases to the intestine is limited and there are no reports on metastases to the rectum . Data frIn conclusion, it is suggested that aggressive, complete surgical resection with radio- and/or chemotherapy is an effective course of treatment for osteosarcomas. This combination therapy can provide a palliative choice and leads to an improved prognosis for patients, even those with metastatic sarcomas.


Ewing sarcoma is a rare tumor that occurs commonly in the long bones of children or adolescents that can also arise in soft tissues including the extremities, retroperitoneum, chest wall, and rarely in the liver as primary sites. We report a case of Ewing sarcoma arising primarily in the liver and, to our knowledge, this is the fourth reported case of Ewing sarcoma occurring in the liver.A 27-year-old Japanese woman was admitted with sudden onset right upper abdominal pain. Clinical examination revealed a multilocular cystic mass consisting of thickened, irregular septa and nodal walls in the right hepatic lobe. Ultrasound-guided aspiration biopsy of the liver mass showed clusters of small atypical round cells and the clinical preoperative diagnosis was mucinous cystadenoma of the liver. The patient underwent an extended right hepatectomy and histopathological findings revealed sheet-like proliferation of small- to medium-sized round cells. Tumor cells were positive for periodic acid-Schiff reaction and immunoreactive for glycoprotein C99 and gene NKX2.2, as well as the neuroendocrine markers, CD56 and synaptophysin. EWS-FLI-1 fusion transcript type 1 was detected by reverse transcriptase polymerase chain reaction. Pathological and molecular analysis confirmed the diagnosis of Ewing sarcoma arising primarily in the liver and the patient received adjuvant systemic chemotherapy with vincristine, doxorubicin, and cyclophosphamide, alternating with ifosfamide and etoposide. We found no evidence of recurrence 15 months after completing chemotherapy.We present an extremely rare case of Ewing sarcoma arising primarily in the liver. To our knowledge, this is the fourth reported case of Ewing sarcoma occurring in the liver, and the first case with a multilocular cystic liver mass. Imaging examinations of the other three reported cases showed solid tumors and a diffuse enlarged liver without mass lesion. Clinicians should consider the possibility of Ewing sarcoma in young patients with a multilocular cystic mass with thick and/or irregular cyst walls in the liver. The Ewing sarcoma family of tumors (EFTs) is a rare entity that includes extraosseous Ewing sarcoma (ES), primitive neuroectodermal tumor (PNET), Askin tumor, and atypical ES. ES represents the second most common primary bone malignancy of children and young adults  and most3/μL) with normal transaminase levels and negative hepatitis B surface antigen and antibody. Serum antibodies for hepatitis C virus, human immunodeficiency virus, and Entamoeba histolytica were negative. Levels of tumor markers of carcinoembryonic antigen and carbohydrate antigen 19–9 were within normal limits.A 27-year-old previously healthy woman was transferred to the emergency department of another hospital in June 2012 because of sudden onset right upper abdominal pain radiating to her back. She was afebrile and other vital signs were normal. Physical examination revealed no abnormality except for right upper abdominal tenderness. Enhanced computed tomography (CT) scan revealed a multilocular cystic mass with enhanced septa, measuring 82 × 66 mm in liver segment 8 , retroperitoneum or intra-abdominal tissues (14%), viscera (8%), and other sites (21%) . Viscera%, retropTo our knowledge, only three published case reports discuss ES arising in the liver –8 and beCystic liver lesions generally represent a heterogeneous group of diseases. Simple solitary cyst, polycystic disease, parasitic cyst, neoplastic (primary or metastatic) disease, duct related disease, false cyst, and ciliated foregut cysts are classified as hepatic cysts . AlthougES is described histologically as small, blue, round cells with hyperchromatic nuclei and scant cytoplasm. The morphological appearance of ES is similar to that of other small, blue, round cell tumors, including lymphoma, mesenchymal chondrosarcoma, medulloblastoma, desmoplastic small round cell tumors, and rhabdomyosarcoma. PAS stain is usually positive for ES because of the presence of abundant glycogen  and the The standard treatment for extraosseous ES is not established because of its rarity; therefore, we used the same strategy as for osseous ES . Modern To our knowledge, this is the fourth reported case of Ewing sarcoma occurring in the liver, and the first case with a multilocular cystic liver mass. Imaging examinations of the other three reported cases showed solid tumors and a diffusely enlarged liver without a mass lesion. Clinicians should consider the possibility of ES in young patients with a multilocular cystic liver mass with thick and/or irregular cyst walls.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.


Salivary gland choristoma (SGCh) is defined as the presence of normal salivary tissue in an abnormal location. It is a rare entity in oral and maxillofacial region and its ethiology is unknown. The typical presentation of salivary gland heterotopia is an asymptomatic mass that may or may not produce saliva. Some examples of ectopic salivary tissue in the pituitary gland, in the lymph nodes, in the middle ear, in the neck, in the jaw, in the thyroid gland, in the mediastinum and in the rectum have been documented in literature.We report the case of a 61-year-old male presented with a bilateral tumorlike mass in the cheek. The mass was painless, of fibrous consistency and had size change with time. The histological diagnosis was salivary gland choristoma in the buccinator muscle. In this article, we will revise the characteristic of salivary gland heterotopias and we present a report case that has not been described in literature: a bilateral choristoma of salivary gland in the buccinator muscle, which should be included in the differential diagnosis of head and neck masses. Key words:Choristoma, heterotopias, ectopic tissue, salivary gland, buccinator muscle.  It is also denominated as l tissue . It is dSGCh has been documented in several areas of the body but rarely in the orofacial region ,2. The mSeveral different heterotopias as osseous choristoma, cartilaginous choristoma, gastric mucosal choristoma, glial choristoma and thyroid choristoma have been described in oral and maxillofacial region .The etiopathogenesis of this entity remains uncertain although it is believed that it is due to a developmental anomaly . Sex andThe present case is the first described in the literature of a salivary gland choristoma in the buccinator muscle.A 61-year-old man was referred to the Unit of Oral Surgery of the University of Barcelona in January of 2010 presenting a bilateral tumefaction in the genian region. The patient had a chronic gastritis and reflux gastritis treated by metoclopramide since 1990 and a C-reactive protein level increase in his last blood analysis. He had seen treated of a depression since 1995. He didn’t have either allergies or toxic habits. He had undergone amigdalectomy (1967) and appendicectomy (1972).The patient had a bilateral mass in both cheeks, rounded, 1.5 cm of diameter, painless, of fibrous consistency, adhering to deep structures, without erythema and with a reported evolution of 2 years. The most striking clinical feature was sporadic swelling, associated sometimes with food intake. The swelling developed in 5-7 days and remained from 15 days to some months. The patient visited an Oral and Maxillofacial Surgery service a year ago and was treated with antihistaminics and muscular relaxants that were unsuccessful to solve his complain.A magnetic resonance of middle and lower face with axial planes was informed as unspecific pathology of the buccinator muscle Fig. .An incisional biopsy was perfomed with local anesthesia and intraoral approach. The incision was 2 cm in length and reached the buccinator muscle where the lesion was located, and two samples of 0.5 cm of diameter were taken , fibrosis and unspecific chronic inflammation Fig. .Three months after the operation the right cheek experienced some swelling (approximately 1 cm increase in diameter). We explained to the patient the benign nature of the lesion and he decided not to have it removed.The criteria for diagnosis of choristoma are a tumorlike growth in the soft tissues or an osseous radiolucency in a radiograph, histologically an ectopic tissue with a normal pattern, with no neoplastic features and topographically a tissue’s type not normally found in the region . SGCh haet al. explain that many lesions in this series were noted at birth or early in childhood and the late diagnosis were when the size was very small or there was an absence of symptoms is nonexistent (In literature, it has been reported that the age of diagnosis of this entity is variable, between birth to 81 years old ,8,12,13.existent . There iexistent ,12,13.The presentation of bilateral and symmetric SGCh, just as our case, is very rare. However, few cases of bilateral choristomas in the neck have been described in literature ,7.The most frequent clinical presentation is a painless tumor . This maIt must be taken into account that the clinical presentation depends of location. SGCh in the jaw, called Stafne bone cavity, are usually radiological findings ,14. Neveet al. (Aetiopathogenesis is unknown but it is believed to be related to embrionary development ,2,4,5,13et al.  reportedet al. .The SGCh histology shows normal salival tissue, mucous and mixed acini predilection that may be accompanied with fibrosis and, sometimes, may difficult the histological diagnose. Some of the variation seen within the associated ducts, such as areas of squamous metaplasia, may be related to chronic inflammation and lymph infiltration secondary to physical pressure of retained mucus ,7. The pTumours in SGCh are rare and 80% of them are benign ,8. The mThere is much controversy in the treatment of SGCh because of the malignization possibility. There are two opinions. In one hand, some authors believe that salival heterotopia is a normal tissue and doesn’t require complete excision when it is diagnosed histologically. Choristoma removal is only necessary when there are signs of infection or neoplasm ,13,17. I


Malignant rhabdoid tumors were originally described in children. Subsequently, the same histological pattern was described in adults. Malignant rhabdoid tumors are aggressive neoplasms that have been reported in multiple organs. To our best knowledge, only 16 previous cases of rhabdoid tumor in the colon have been described in the literature. We present the case of an 87-year-old lady who was diagnosed with a rhabdoid tumor of the colon that relapsed rapidly after surgical resection. The literature concerning this unusual neoplasm was subsequently reviewed with comparison of all known cases in the literature. In 1978, Beckwith and Palmer first described Wilms' tumors with “rhabdomyosarcomatoid” features in children. This pattern was characterized by diffuse sheets of polygonal cells with acidophilic cytoplasm and rounded vesicular nuclei , 3. ThesThe patient was an 87-year-old Caucasian female of Russian descent who presented to our hospital with a two-day history of generalized weakness, profuse nonbilious nonbloody vomiting, and decreased oral intake. The patient complained of diffuse abdominal pain but had no change in bowel movement. The patient had been suffering from decreased appetite for weeks. The patient's past medical and surgical history are noncontributory because the patient had not seen a physician nor taken a medication in many years. In the emergency department, vital signs showed blood pressure of 88/54, pulse rate of 115 bpm, a respiratory rate of 20, and a temperature of 98.8 F. Physical examinations showed a frail looking lady with pale conjunctiva and skin, lungs were clear to auscultation bilaterally, and heart auscultation did not elicit any murmur rubs or gallop. Abdominal examination elicited a right upper quadrant mass on deep palpation without associated tenderness or distention and with positive bowel sounds in all quadrants. The rest of the physical exam was unremarkable. A stool guaiac test performed at bedside was negative. On further interrogation of the family, we were informed that the patient was a nonsmoker and occasional alcohol user and had no known drug allergies. The patient had no family history of malignancy.The patient was urgently stabilized in the emergency department with intravenous fluids, transfusion therapy, and antibiotics and was admitted to the intensive care unit for monitoring.Blood tests on admission were as shown in An emergent CT scan of the abdomen and pelvis done without contrast showed a large concentric and enveloping 12.1 × 9.3 × 8.4 cm mass along the hepatic flexure of the transverse colon with associated mass-effect and obstruction along a portion of the distal jejunum and innumerable splenic hypodensities. A chest radiograph done on admission did not show any suspicious nodules. A colonoscopy showed a friable, infiltrative, and ulcerated tumor which occupied 100% of the circumference of the transverse colon and caused a severe 80% obstruction of the lumen. Biopsies were taken. Additional findings included diverticulosis in the sigmoid and descending colon and incidental polyps which were removed. The patient underwent right hemicolectomy, partial omentectomy, small bowel resection with primary anastomosis, and a twelve-lymph node resection. The postoperative course was unremarkable. Macroscopically, the mass was a 12 × 9 × 9 cm necrotic tumor. Pathological findings showed a high grade malignant neoplasm, in favor of undifferentiated carcinoma with rhabdoid features involving ulcerated mucosal and submucosal fragments with extensive necrosis. The tumor was positive for calretinin, pancytokeratin, vimentin, CAM 5.2, and neuron specific enolase which are in favor of the aforementioned diagnosis. The tumor was focally positive for epithelial membrane antigen. One of twelve lymph nodes that were found in the resected specimen was positive for micrometastasis. The tumor extended to the free serosal surface of the right colon and a loop of small bowel with extensive vascular involvement and no perineural involvement. Surgical margins were negative microscopically. The final pathologic stage was pT4B, pN1 (mic), and pMx with microsatellite instability. The initial staging was stage IIIC without ruling out metastasis since the splenic masses were never evaluated with pathology. Upon follow-up with the oncologist and further discussion of the case with the family, a trial of capecitabine was suggested. The family opted not to undergo any treatment but decided to follow with the oncologist closely and manage symptoms.Upon follow-up with the oncologist one month after the initial assessment, a repeat CT of the abdomen showed development of moderate amount of intra-abdominal pelvic ascites and interval development of 6.1 × 3.1 cm heterogenous mass in the right abdominal wall consistent with tumor recurrence associated with new mesenteric, gastrohepatic ligament and retropritoneal adenoapthies. Multiple low-density splenic lesions stable in appearance from the previous study were identified. A repeat CT of the chest showed a 1.2 cm solid nodule in the left lung apex. Physical exam in the clinic elicited effectively a 5 cm nontender palpable mass at the surgical scar. Repeat CEA was 11. The patient passed away two months later from complications of her disease.Colon carcinoma with rhabdoid features is an uncommon pathological entity. It is almost always associated with unfavorable prognosis. The existence of this tumor as an independent pathological entity is still under debate since it is considered by some experts to be merely a phenotypical variation of the tumor . MalignaImaging does not seem to be particularly helpful in diagnosing tumors with rhabdoid features since it does not present any specific radiological findings . The rha SMARCB1 biallelic inactivation in rhabdoid tumors [ SMARCB1  is a core subunit of the SWI/SNF chromatin remodeling complex which regulates chromatin structure and thus plays an important role in gene expression [ SMARCB1 as the only consistently recurrent event [ SMARCB1 alteration [ SMARCB1 mutations strongly predispose to rhabdoid tumors [ SMARCB1 biallelic loss is now used in diagnosing these tumors by detecting loss of protein expression in the nucleus by immunostaining [ SMARCB1 loss detection can thus play an integral part in differentiating rhabdoid tumors from other tumors with similar histologic features [ SMARCB1 loss seem to be related to its effect on p16 INK4a and cyclin D1 which play a significant role in the cell cycle and on RhoA signaling which enhances cell migration [ SMARCB1 loss might provide insight to oncogenesis and may help in developing therapies for a multitude of SWI/SNF mutant neoplasms [ SMARCB1 losses have also been described in other tumors such as renal medullary carcinoma, epithelioid sarcomas, and nerve sheath neoplasms [In 1998, Versteege et al. describedd tumors , 25. SMApression . The inapression . Even thnt event , 29. Up teration . Germlind tumors  and subsstaining . Thus atfeatures . This lofeatures . The oncigration . Understeoplasms . SMARCB1eoplasms .Tumors with rhabdoid features have been described in multiple organs. The rhabdoid feature seems to be associated with poor prognosis. We present the case of an 87-year-old female who was found to have an undifferentiated carcinoma of the colon with rhabdoid features. To our best knowledge, this case is the seventeenth case of a tumor with rhabdoid phenotype to be described in the colon.


To describe the first case of partial vision recovery in a 32-year-old woman with iatrogenic retinal artery occlusion (RAO) following glabella calcium hydroxylapatite (CaHA) injection, and to explore appropriate diagnostic and therapeutic measures according to a literature review.A 32-year-old woman had left eye RAO and a bilateral visual field defect after CaHA injection into the glabella region. Topical and systemic intraocular pressure lowering agents, isovolemic hemodilution, globe massage, and anticoagulation with acetylsalicylic acid were prescribed. Carbogen inhalation and oral corticosteroids were also given. In addition to the above therapies, hyperbaric oxygen therapy (HBOT) was implemented as adjuvant treatment. The final best corrected visual acuity (BCVA) of the left eye improved from hand motion at 15 cm to 0.1. Improved retinal circulation and decreased retinal vessel leakage were found in the follow-up fluorescein angiography. However, there were still multiple emboli in the conjunctival and retinal arteries.This is the first case report on partial recovery of BCVA after iatrogenic RAO following cosmetic CaHA injection. Because no reliable treatments have been reported for such complications, HBOT may be considered as an alternative adjuvant therapy. As soft-tissue augmentation of the face has become more common, there has been an increasing number of cases of ocular complications after facial filler injections, including hypersensitivity reactions, ecchymosis, anterior chamber inflammation, strabismus, ptosis, ophthalmoplegia, and retinal vessel occlusion. Among tA 32-year-old woman sought treatment in the emergency department reporting sudden vision loss in her left eye. Four hours previously, she received vaginal plastic surgery and CaHA filler injection into the glabella region by a local plastic surgeon under general anesthesia. After waking up, she noted vision loss in her left eye, and was immediately transferred to our hospital. The initial best-corrected visual acuity (BCVA) at our emergency department was hand motion at 15 cm in the left eye and 1.0 in the right. The left pupil was dilated with a positive relative afferent papillary defect. There was neither ophthalmoplegia nor strabismus. Slit lamp examination showed multiple emboli along the conjunctival vessels Figure A, and flDespite the treatment, progressive blurred vision was still found in the following few hours. Intra-arterial fibrinolysis using recombinant tissue plasminogen activator was not performed because of higher rates of adverse side effects. An alteOur literature review revealed that a retrograde embolic mechanism is generally thought to be the pathogenesis of RAO after cosmetic facial filler injections, 6. OnceOur case is the first case report of vision and circulation improvement in posterior segment ischemia due to CaHA filler injections. A variety of measures have been advocated in an attempt to treat non-arteritic retinal artery obstruction, but there were no generally agreed upon treatment regimens, with no suggested treatment regimens for iatrogenic RAO. In 2010, Sung et al.. The maiIn our literature review of 46 case reports, 15 reported therapies for iatrogenic RAO, including dilation of the artery by breathing carbon dioxide/carbogen and usinThe major difference in our case from previous reports is the early HBOT within 24 hours after the onset of symptoms. A number of investigations have shown that early HBOT within 24 hours after the onset of symptoms had beneficial effects for visual acuity improvements in eyes with RAO, and there were no serious major complications–34. HoweAnother issue that needs to be mentioned is the possibility of central nervous system infarction. Just as the injected materials can be pushed into the ophthalmic artery, an excessive force may also cause retrograde emboli into the internal carotid artery, resulting in a cerebrovascular event. In our review, 7 of the 46 cases 7/46, 15.2%)[.2%15, , 30, 36 In conclusion, considering there are no reported reliable treatments for iatrogenic RAO, a poor visual acuity outcome is inevitable. In this report, we offer our experience with HBOT for the treatment of iatrogenic RAO. We found that early HBOT had some beneficial effects on retinal perfusion and visual acuity, while there were no major complications. Therefore, along with recommended treatments by the EAGLE study, HBOT seeWritten 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.


Purpose: The aim of this work is to report a review of the literature concerning epidemiology, clinical and radiographic features as well as treatment of odontogenic myxofibroma (MF). Methods: The PubMed database was searched using the following keywords: “odontogenic myxofibroma”, “odontogenic fibromyxoma”, “myxofibroma of the jaw” and “fibromyxoma of the jaw”. Results: Fifteen articles reporting the experience with 24 patients were identified. Male/female ratio was 1:1.4 and the average age was 29.5 years. The most frequent location was the mandible. In 66.7% of the cases the radiographic appearance was a multilocular radiolucency. Swelling was observed in 13 patients (92.86%), varying degrees of pain in 5 (35.71%) and paresthesia in only one patient (7.14%). Six out of 24 patients (26.09%) were treated with radical surgery and 17 (73.91%) with a conservative approach. In two out of 21 cases (9.52%) a recurrence was reported.Conclusions: MF is an extremely rare tumor and no agreement exists on the causes of its development. According to the present review, the choice of treatment should depend on variables such as localization, presence of a primary or of a recurrent lesion, age, general medical conditions and aesthetic needs of the patient. Key words:Odontogenic myxofibroma, myxofibroma of the jaw, odontogenic tumors, oral surgery, oral pathology. Myxofibroma (MF) of the jaws is a rare, benign, odontogenic tumor of possible mesenchimal origin firstly described by Virchow in 1863 , 10 were males (41.7%) .The youngest patient was 8 years old; the oldest was 71. Average age was 29,5 years.Thirteen tumors (54.2%) were located in the mandible, ten of these (76.9%) affecting the posterior area. Eleven MFs (45.8%) were located in the maxilla, 6 (54.5%) in the anterior and 5 (45.5%) in the posterior area .The radiographic appearances are reported in The majority of cases  appeared as a multilocular (“tennis racket” or “honeycomb”) radiolucency; less frequently  MFs presented a unilocular radiographic pattern Fig. .Most of the Authors reported an histologic pattern consisting of abundant myxoid connective stroma, areas of moderately dense collagen fibers, and strands of odontogenic epithelium within the connective tissue stroma . The tumThe range of follow-up varied from 6 months to 4 years. In two out of 21 cases (9.52%) a recurrence was reported , terms such as “soap-bubbles”, “ground-glass” or “tennis racquet strings” have all been used to describe the radiographic appearances of these lesions. As these descriptors may be variably interpreted and somewhat confusing, a more precise definition of the radiological features of myxomas and MFs may be warranted in an attempt to establish more objective diagnostic criteria.According to the literature, clinical and radiographic differential diagnoses of MFs include ameloblastoma, ameloblastic fibroma, odontogenic fibroma, odontogenic keratocystic tumor, central hemangioma, aneurysmal bone cyst and other rare entities like desmoplastic fibroma. The gross appearance of the surgical specimen which displayed a hard-fibrous consistence, could lead the surgeons toward the hypothesis of a myxomatous lesion .As for the other odontogenic tumors, definitive diagnosis of MFs is based on histopathologic evaluation. In case of big lesions, a biopsy may be necessary to establish the nature of the tumor and to plan the therapeutic approach. MF is not radiosensitive and the surgical excision has been reported as the treatment of choice . No agreRecurrence is probably associated to local invasion into cancellous bone beyond radiographically visible margins in absence of tumor encapsulation. Recurrence rate can apparently be reduced with a more aggressive treatment by performing a partial or complete segmental bone resection with tumor free margins of 1.5 cm. Such a treatment can be preferred in the maxilla, where the closeness to the maxillary sinus, the zygoma and the lower part of the orbital cavity can be a critical factor in case of recurrence .The choice of conservative surgery is also supported by the absence of evidence of malignant transformation of MFs as well as the low recurrence rate reported in some case series after conservative treatment .The patient should be monitored for at least three years after the surgical intervention as the recurrence rate seems to be higher during this period .In conclusion, the choice of treatment mainly depends on variables such as mandibular or maxillary localization, presence of a primary or recurrent lesion, age, general medical conditions and aesthetic needs of the patient.


Fibrolamellar carcinoma (FLC) is an uncommon form of primary liver malignancy with unique clinical, histological, and biological characteristics. It is usually seen in young adults without underlying liver disease. Histologically, it shows large cells with abundant eosinophilic cytoplasm, large vesicular nuclei, prominent nucleoli, and lamellar type fibrosis. In contrast, classical hepatocellular carcinoma (HCC) is typically present in elderly male patients with cirrhosis. It is the most common histological subtype, and it is characterized by its resemblance to the normal liver, both in its growth pattern and its cytology. The unusual case of a liver carcinoma that presented with histological features of both FLC and classical HCC is herein reported. This was the case of a 37-year-old female complaining of diffuse abdominal discomfort and epigastric pain for two months. She was referred to us for further management after she was diagnosed with HCC in a noncirrhotic liver. She underwent a left-sided hepatectomy. A yellow nodular mass with well-defined borders and a necrotic center was present in the resection specimen. The morphological features and immunohistochemical studies were consistent with a diagnosis of FLC mixed with classical HCC. The patient was followed up for five months, and no signs of recurrence were evident. Fibrolamellar carcinoma (FLC) is a rare variant of primary liver malignancy with unique clinical, histological, and biological characteristics. It was first reported as a separate entity by Hugh Edmondson in 1956, who described the typical gross and microscopic features of this neoplasm as “a liver cell carcinoma in which stroma is profuse and the similarity of the cancer cells to normal cells is striking” .FLC accounts for up to 8% of all HCC, occurs predominantly in younger individuals, has no gender predilection, and develops in the absence of underlying liver disease –5. It isTo our knowledge, only three cases of a liver carcinoma with elements of both classical HCC and FLC intimately mixed within one mass have been reported , 7. Two A 37-year-old female presented to our clinic with a two-month history of diffuse abdominal discomfort and epigastric pain. The abdominal ultrasound showed a 9.6 cm oval-shaped, well-demarcated, thin-walled, heterogeneous mass located in the left lateral segment of the liver (segments II and III). CT and MRI scans revealed a well-defined, encapsulated, nonenhancing mass with a central scar, surrounded by normal liver parenchyma. The radiologic diagnosis suggested a FLC in a noncirrhotic liver. Laboratory tests showed abnormally elevated transaminases. Serum tumor markers, including a-fetoprotein (AFP) and carcinoembryonic antigen (CEA), were within normal limits. Viral hepatitis and autoimmune markers were negative. A biopsy was performed at an outside institution before referral, and a diagnosis of classical HCC with a trabecular pattern in a noncirrhotic liver was rendered. The patient's past medical history was unremarkable, and she had no risk factors for chronic liver disease. Physical examination revealed a painful mass that comprised the upper abdomen.The patient underwent an uneventful open left-lateral hepatectomy  and was Macroscopic examination of the resected specimen revealed a multinodular, well-circumscribed, and partially encapsulated mass that measured 13 cm in greatest dimension. Cut surface showed a variegated appearance with some yellow discoloration and central necrosis. The surrounding liver parenchyma had a normal appearance and no evidence of fibrosis or nodularity was seen. The mass was located 3 cm from the surgical margin, and no other lesions were noted.Microscopically, the tumor had two different cell populations, one of which showed large, polygonal cells with eosinophilic cytoplasm, large nuclei, prominent nucleoli, and rare mitoses. In addition, there were scattered cytoplasmic inclusions with a ground-glass appearance, known as pale bodies. The tumor cells were surrounded by a fibrolamellar type stroma. These findings were consistent with FLC.Adjacent to these areas, a second group of cells with marked cytologic atypia, nuclear pleomorphism, hyperchromatic nuclei, intracytoplasmic Mallory's hyaline and eosinophilic globules, and zones of necrosis was observed. Morphologically, these areas were consistent with classical HCC with a trabecular pattern. Vascular invasion and tumor thrombi were also present. The background liver showed no evidence of cirrhosis .Immunohistochemical studies showed both components to be reactive for hepatocyte (Hepar-1) and to focally express AFP. Endothelial wrapping was highlighted with CD34, and a canalicular staining pattern was observed with polyclonal CEA. Both groups of neoplastic cells were positive for CK7 and negative for CK19. A diagnosis of a hepatocellular carcinoma with both FLC and classical HCC features was rendered.We present a case of a liver tumor with unusual morphology and features of both classical HCC and FLC in a patient with no predisposing factors. FLC is a malignant hepatocellular tumor with distinct clinical and pathologic characteristics that differ from classical HCC, predominantly in FLC there is no consistent mutation, and shows fewer chromosomal abnormalities compared with HCC .To our knowledge, there are only five cases reporting the presence of classical HCC coexistent with FLC in the literature . Two casFLCs have a predilection for the left lobe of the liver, and grossly, they tend to be yellow to pale tan and firm , 11, 12.Microscopically, the defining features of FLCs are large polygonal cells with abundant eosinophilic (oncocytic) cytoplasm, large vesicular nuclei, large nucleoli, and lamellar type fibrosis , 11, 12.On the other hand, classical HCC is usually nodular with or without a fibrous capsule, and it is softer than the normal surrounding liver. It varies in color and can be yellow, green, brown, or heterogeneous .Histologically, the main hallmark of classical HCC is its similarity to the normal liver both in its plate-like growth and its cytology . It is cImmunohistochemically, classical HCC is characterized by cytoplasmic positivity for Hepar-1 (approximately 90% of the cases), patchy AFP, fibrinogen, and cytokeratins 8 and 18. Polyclonal CEA and CD10 show canalicular patterns of positivity. Special stain for reticulin shows patchy or diffuse loss of the reticulin network . CD34 hiIn the present case, the patient had no evidence of underlying liver disease, she had no risk factors for HCC, AFP levels were within normal limits, HBV titers were negative, and imaging showed a mass in the left lobe of the liver, all of which are characteristic clinical features of FLC. In contrast, she presented with vague, nonspecific abdominal signs and symptoms, and with mild elevation of the liver function tests, which may be seen in both types of tumors. The age at presentation of the disease (37 years old) is not usual for either classical HCC or FLC.Microscopically, the tumor showed distinct areas in which the morphological elements of classical HCC and FLC were mixed. However, the immunoreactivity for CK7, which was expected to be expressed solely by the FLC component, was positive in both groups of neoplastic cells. It should be noted that some ≤30%) classical HCCs may be reactive for CK7, in particular those in children and young adults 0% classi. In this DNAJB1-PRKACA chimeric transcript that is missed in the domain that binds the regulatory subunits of PKA and may represent a diagnostic marker in FLC; nonetheless further research has to be done to define it as a therapeutic target [Currently there are no molecular diagnostic tests for FLC; previous studies detected cAMP-dependent protein kinase A (PKA) in peripheral blood as a universal serum biomarker for cancers of various cell types . The DNAc target .We did not find any risk factors or environmental exposure in the patient's history that could explain the etiology of this neoplasm. As a matter of fact the surrounding liver parenchyma was normal and did not have any signs of cirrhosis or underlying liver disease.In summary, we hope that our report will increase awareness of this combined entity in the liver and that further experience contributes in the establishment of prompt and accurate diagnoses involving not only proteins expression through immunohistochemistry but also genetic studies in future cases.


The urachus establishes a connection between the dome of the bladder and the umbilicus throughout fetal life. If the urachus does not close completely, malignancy is a potential complication. The primary treatment for malignant urachal tumor is surgical excision. A 61-year-old male patient diagnosed with urachal carcinoma had undergone partial cystectomy 25 years previously. Twenty years later, local recurrence was treated with another partial cystectomy without umbilical remnant excision. Recurrence at the umbilical site was excised 2 years later, but intraperitoneal invasion had occurred, and the patient underwent a total colectomy at that time. Local disease and disseminated metastases in the thorax and intra- and extraperitoneal areas were noted upon admission to our hospital. Urachal carcinomas are usually aggressive tumors, and surgical treatment should include partial or radical cystectomy and excision of the urachus and umbilicus, to prevent local recurrence and distant metastasis. The urachus is the vestigial remnant of the cloaca and the allantois, forming a fibrous embryonic remnant that extends from the dome of the bladder to the umbilicus. Urachal remnants may be noted in infants and during childhood, where they are most frequently found as thin-walled, internally homogenous cysts, containing straw yellow umbilical fluid. The development of malignancy is noted in adults. Urachal carcinomas constitute about 0.2% of all bladder tumors . The priHerein we reported an unusual case of a patient with an inadequately treated urachal carcinoma who did not receive chemotherapy or radiotherapy, over a period of 25 years.A 61-year-old male was admitted to our urology department with complaints of abdominal pain, hematuria, a known mass in the abdomen, and discharge of fluid from the umbilicus. He had been evaluated 25 years ago for macroscopic hematuria, and an urachal tumor was identified. Transurethral resection of the bladder tumor (TUR-BT) revealed mucinous adenocarcinoma of the urachus, and a partial cystectomy was performed. After the operation, the patient was noncompliant with the recommended follow-up protocol. Twenty years later, he was examined by a urologist for the same complaint. Cystoscopy and TUR-BT confirmed the same diagnosis. Thoracoabdominal computed tomography (CT) verified a local recurrence of urachal carcinoma, but revealed no distant metastases. Radical cystectomy was recommended to the patient, but he did not consent. The patient underwent a second partial cystectomy at another hospital 6 months later but the umbilical remnant was not resected at that time. Three months later, local recurrence was identified, and local excision of both tumor and umbilicus was performed. Two years later, intraperitoneal invasion had occurred and the patient underwent a total colectomy. All laboratory findings were normal except for hematuria. In radiological investigation, a CT scan revealed a massive lesion filling the bladder lumen with extension to the anterior wall of the abdomen, a heterogeneous internal structure, and amorphous punctate calcifications . The lesThe patient was deemed to be inoperable and chemotherapy was subsequently initiated. The patient ultimately was treated symptomatically, as he was unable to tolerate the chemotherapy, and died 6 months later.Urachal diseases may be congenital or acquired . While tThe development of malignancy in urachal remnants is typically seen in or after middle age . ClinicaUltrasonography is the first choice for imaging in the diagnosis of urachal malignancy, because of its widespread availability, ease of use, and inherent suitability in the evaluation of cystic lesions. Uncomplicated cases may often be diagnosed with routine abdominal ultrasound alone. Such malignancies may be found on the anterosuperior surface of the bladder, with midline placement, and appear elliptic and hypoechoic in structure. However, cystography or sinography may often be required for final diagnosis .CT and magnetic resonance imaging (MRI) are beneficial in monitoring tumor progression, identifying both extension and spreading, as well as for preoperative surgical planning. However, in the case of complicated cystic lesions or contrast-enhancing lesions, it may be extremely difficult to differentiate between infected urachal remnants and urachal carcinomas. Cysts with mural nodularity and calcification in contrast-enhanced computed tomography (CECT), as well as hematuria and the absence of findings of infection in the perilesional soft tissues, are usually signs of malignancy . HoweverDespite the fact that a normal urachus is wrapped in transitional epithelium, urachal carcinomas occur as adenocarcinomas in roughly 90% of cases . ConsistIn the case of urachal carcinoma, there is no consensus over the implementation of partial cystectomy of the dome of the bladder versus radical cystectomy; for both there is a consensus on the removal of urachus and umbilicus , 7. CarcBruins et al. stated that the peritoneum is the location where distant metastasis is most commonly seen, starting at the age of approximately 45 years . While iUrachal carcinomas are usually aggressive tumors but may also present as slow growing and locally recurrent. Therefore, correct radiological assessment of patients is important. Surgical treatment should include partial or radical cystectomy, as well as excision of the urachus and umbilicus to prevent local recurrence and distant metastasis.


Ocular rhinosporidiosis is a chronic granulomatous infection caused by a newly classified organism that is neither a fungus nor bacterium. It often presents as a benign conjunctival tumour but may mimic other ocular conditions. It is most often described in India. In Africa cases have been reported from South Africa, Kenya, Tanzania, Malawi, Uganda, Congo and Ivory Coast.A 54 year old man was seen in Kenya with a lesion that resembled a conjunctival papilloma. We report resemblance to conjunctival papilloma and the result of vital staining with 0.05% Toluidine Blue.Ocular rhinosporidiosis occurs in East Africa. It may resemble conjunctival squamous papilloma. Vital staining with 0.05% Toluidine blue dye did not distinguish the two lesions well. Rhinosporidium seeberi[Rhinosporidiosis is a chronic granulomatous infection of the mucous membranes  caused by m seeberi. This ism seeberi. CurrentRhinosporidiosis affects both adults and children and is commonly seen in otolaryngology. The largest reported case series of rhinosporidiosis consisting of 462 cases in India found that the disease mainly occurs in the nose and nasopharynx (81.1%) while the eye was affected in 14.2% . AnotherOcular rhinosporidiosis affecting the conjunctiva was first described in India in 1912 . CurrentHow the disease is acquired remains an enigma. Rhinosporidiosis has been associated with migrants from endemic areas -16. AlthOcular rhinosporidium most often presents as a polypoid mass of the palpebral conjunctiva . It may In a series of 63 cases from India that included nasal, nasopharyngeal and ocular disease, routine haematology tests did not show any abnormality and while cytology of smears obtained via fine needle aspiration or cytology has a role in diagnosis, the mainstay remains histology . Vital sRhinosporidium seeberii but no human trials have been reported [The treatment is surgical excision with or without cautery at the base and recurrence is described as rare . Scleralreported . Human areported .A 54-year-old male presented to the eye clinic at the PCEA Kikuyu Hospital on the outskirts of Nairobi with a 16 month history of a painless lump on the surface of the right eye. Concerned about the appearance, he attributed the lesion to a foreign body that entered that eye while he was trimming a hedge.No other family member or neighbour had a similar disease. Social history included living in Homa Bay district on the shores of Lake Victoria from birth to 18 years age, then Kapsabet, a highland area in the Rift Valley until the age of 26 years, followed by Nairobi. He had resided in a low-income area of Nairobi for the past 11 years. Occupational history included working as a gardener for the last 10 years and a cook for 5 years prior to that. Although he grew up in a lakeside area, he had not dived or swum in stagnant water in the recent past.On examination he had a pedunculated 6×11 mm wide fleshy mass at the medial canthus of the right eye (Figure Histological analysis revealed multiple sporangia in the conjunctival stroma, an ulcerated squamous epithelium covered by a fibrin plaque whose underlying tissue showed granulomatous tissue, mixed inflammatory cells with lymphocytes showing a maturation spectrum and numerous thick walled sporangia filled with nucleated basophilic endoconidia Figures , 5 and 6There was no recurrence 6 months after excision was performed Figure .Ocular rhinosporidiosis occurs in East Africa. It may resemble conjunctival squamous papilloma. Although toluidine blue has been used as a vital stain of conjunctival lesions, in this case, it was unable to distinguish between an infective and neoplastic cause.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.The authors declare that they have no competing interests.SG first evaluated the case, took clinical photographs and conceived the report idea. EM performed the excision surgery. TO performed the histopathological assessment and made the diagnosis. JK reviewed the case on follow up. AMZ coordinated patient and institutional consent for publication of this clinical material. MSS and MJB evaluated the clinical and histopathology photographs. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2415/14/45/prepub


Solitary fibrous tumors (SFTs) may occur at any site in the body. SFTs can only be conclusively diagnosed based on histopathologic and immunohistochemical characteristics of the tumor. The presence of SFTs in the abdomen and pelvis is extremely rare. To our knowledge no cases of urethral solitary fibrous tumor in the literature have been reported so far. We present a case of a solitary fibrous tumor arising from the urethra in a twenty-three-year-old female presenting with vaginal mass. A 23-year-old sexually active female with no significant medical or surgical history initially presented to colorectal surgery for evaluation of a potential perianal mass. The patient reports having limited medical care and no recent gynecological visits. Three years prior to presentation, the patient first noticed a small growth on her perineum. Over time this growth had increased in size, but it had not interfered with any of her activities. She was still able to have sex and urinate without difficulty. The patient had no reported history of sexually transmitted infections. She denied significant pain, discharge, or bleeding from this mass.During the initial physical examination by the colorectal surgeon it was determined that this lesion was not arising from the perianal region but was in fact of periurethral or anterior vaginal wall origin . The patThe patient was scheduled for an examination under anesthesia, cystoscopy, and planned vaginal excision of the mass from the posterior urethral edge. Further imaging with ultrasound, CT scan, or MRI was deferred pending final pathologic findings.At the time of a surgery, cystourethroscopy showed no evidence of urethral involvement with the mass. The bladder neck and bladder itself were noted to be unremarkable with no evidence of tumor, stones, or diverticulum. After completion of cystourethroscopy, a foley catheter was placed, and the mass was placed on traction. The mass was noted to have a broad base spanning from the 4 o'clock to 8 o'clock position encompassing the anterior vagina and posterior lip of the urethra . The basA frozen section was performed intraoperatively. The pathologist noted benign cytological features with no obvious source. The cut margin appeared free of any involvement. At the end of the case, the patient was transferred to the recovery room in stable condition with foley catheter and vaginal packing in place. Both the foley and vaginal packing were removed prior to the patient being discharged home. The mass measured 5.6 cm in maximal dimension. The mass was polypoid and pedunculated with a smooth and glistening external surface. A section of skin was recognizable. The cut section was homogenous with a tan/white color. The microscopic sections revealed a well circumscribed, unencapsulated mesenchymal neoplasm composed of bland spindle cells with elongated nuclei and indistinct cytoplasm. The cells were arranged randomly with hyper- and hypocellular areas (patternless pattern), in a collagenized matrix with variably prominent, often staghorn-shaped blood vessels . Focal cThat patient presented for a two-week postoperative appointment with urologist with no acute complaints. She was recovering well from her procedure. At her follow-up appointment final pathology results were as follows: Immunohistochemically, the spindle cells were positive for CD34, bcl2, and CD99  and negative for S-100, desmin, and smooth muscle actin. These findings are consistent with a solitary fibrous tumor of the urethra.Periurethral masses are rare, occurring in only 3-4% of all patients . It can Solitary fibrous tumors were first described in 1931. These tumors are of fibroblastic or myofibroblastic origin . SolitarExtrapleural SFTs are observed in middle aged adults, typically between 20 and 70 years of age, with both sexes being affected equally . ReportsSFTs can only be conclusively diagnosed based on histopathologic and immunohistochemical characteristics of the tumor. SFTs are classically described as having a “patternless pattern.” Microscopic examination shows spindle cells dispersed among dense collagen fibers in a storiform arrangement . Tumor cMost of these tumors are asymptomatic unless they grow to cause compressive or obstructive symptoms. In our case, the patient was asymptomatic but the size of the tumor grew to be bothersome to her daily activities. Rarely, these tumors can be associated with Doege-Potter syndrome. This is a syndrome in which the tumor is associated with hypoglycemia, which occurs with less than 5% of all SFTs [SFTs are reported to be malignant in 10–15% of cases . The ratSurgical removal of a periurethral or urethral mass can result in surgical complications. One of the most concerning complications results from scarring of the urethra. This can cause a urethral stricture resulting in bladder outlet obstruction. Bladder outlet obstruction occurs in 2–29% of all women . This obA PubMed search did not yield any reports of urethral solitary fibrous tumor in the literature. There have been several case reports of SFTs arising in other areas of the urogenital tract and female genital tract including the kidney, bladder, vulva, and vagina. Although uncommon, solitary fibrous tumors should be in the differential diagnosis for any soft tissue mass arising from the urogenital tract.


To the best of our knowledge, en bloc left hepatectomy with resection of the tumor in his abdominal wall. A pathological examination of the resected specimen confirmed the diagnosis of hepatocellular carcinoma involving the abdominal wall. Disease-free margins of resection were achieved. Our patient’s postoperative course was uneventful. Eight months after the last surgery, our patient died owing to recurrence and distal metastasis.In September 2012, a 64-year-old Chinese male patient was referred to our hospital because of primary hepatocellular carcinoma located in Couinaud’s segments III and V. Our patient first had a hepatectomy of the liver. Ten months later, he presented with an abdominal wall mass and upper abdominal pain. Computed tomography and magnetic resonance imaging scans demonstrated a 10cm tumor in his left liver with extrahepatic metastases in his abdominal wall. It was determined that he had recurrent hepatocellular carcinoma associated with direct invasion into his abdominal wall. He had an Direct invasion of hepatocellular carcinoma into the abdominal wall is rarely encountered. Complete surgical resection should be considered in patients with an appropriate hepatic functional reserve, with consideration of the technical difficulty relating to tumor involvement with surrounding tissues. Hepatocellular carcinoma directly invading the abdominal wall is rarely reported , 2. To tA 64-year-old Han Chinese male patient presented with primary multi-HCC. He had tumors located in Couinaud’s segments III and V, which were treated with local resection. After surgery, he received transcatheter arterial chemoembolization (TACE). After 10 months, he was referred to our hospital in September 2012 with right upper abdominal pain and an abdominal wall mass , local gauze packing was applied to avoid postoperative bleeding. The operation lasted 2 h. The intra-operative blood loss was 1200mL with six units of red blood cells and four units of fresh plasma transfused during the operation. Continuous abdominal double cannula lavage and low negative pressure drainage were used to drain his abdominal cavity. The abdominal drain and gauze packing were loosened and removed on the third and fifth day after the operation respectively.His postoperative course was uneventful. There were no postoperative complications. Histologic findings were consistent with moderately differentiated HCC invading the abdominal wall. Our patient had a recurrence six months after the second surgery, refused treatment, and died two months later (Figure The second surgery was performed in July 2013. An incision was made between the original incisions, over a colored mass in the abdominal wall (Figure r Figure .Figure 3et al. reported [et al. [Tumor recurrence rates in HCC remain high, at 50% to 60% –15, and reported  that a c [et al.  state th [et al. . TherefoA second surgery is difficult for intrahepatic recurrence and extrahepatic metastasis. Repeat hepatic resection is more difficult than primary resection because of the impaired liver function due to the progression of hepatitis, the presence of adhesions, and modifications in the anatomy caused by the previous operation . In our In this case we had to carefully consider the design of the incision for the second surgery. Tumors on an incision site will grow quickly and rupture, so surgery is necessary. Sometimes the incision will need a skin flap or graft, particularly if the abdominal wall mass is large and a large defect in the incision is left post-surgery. If a skin flap is not sufficient, reconstruction with further skin replacement for the abdominal wall is required. In our patient, after combined resection of the intrahepatic recurrence and abdominal wall mass, his abdominal tension was reduced. This allowed us to suture the incision so a flap was not required.We selected to treat our patient’s extrahepatic tumor invasion of the abdominal wall and HCC recurrence with combined resection. Our experience may be useful in improving the cure and resection rates for liver cancer that is unresectable. We have reported a rare case of a patient who successfully underwent repeat resections for hepatic recurrences directly invading his abdominal wall after hepatectomy for HCC. To prevent postoperative recurrence of HCC, patients require comprehensive treatment.Written informed consent was obtained from the patient’s next of kin 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.


No recurrence and metastasis were observed in the patient during the subsequent 25-month follow-up period.A 31-year-old male patient with a six-year history of left inguinoscrotal swelling was admitted to the Second Xiangya Hospital . The mass was not found to be associated with intraperitoneal pressure. Ultrasonography and computed tomography examinations demonstrated several solid, botryoidal masses involving the spermatic cord, with limited capacity of mobility. The demarcation between the masses and the left testicle was clear; thus, the masses were removed by a left spermatic cord tumor resection via a left inguinal approach, under epidural anesthesia. Positive staining of the tumor markers, CD34 Solitary fibrous tumors (SFTs) are a rare type of mesenchymal tumor, which were first described by Klemperer and Rabin in 1931 . HoweverDue to the rarity of these tumors, there was no consensus on its standard treatment. However, treatment for SFTs include surgical resection, radiotherapy and chemotherapy. Resection of the tumor with a sufficient surgical margin was the mainstay of treatment. However, a number of studies have demonstrated that radiotherapy and chemotherapy, with ifosfamide and adriamycin, is effective ,8. HowevThe patient was a 31-year-old male with a six-year history of left inguinoscrotal swelling, which had been gradually increasing in size for the past three years. The size of the swelling showed no association with increased intraperitoneal pressure. Physical examination revealed no evident defects in the appearance of the scrotum, with the exception of several hard, botryoidal masses, located between the left testicle and the inguinal region, with smooth surfaces and limited capacity of mobility. Ultrasound examination revealed several solid, well-demarcated, hypoechoic extratesticular masses arising from the left spermatic cord, the largest of which was ~3 × 2 cm in size. The masses were separated from the cutis and showed no connections with the left testis and epididymis, and no vascularity was observed. The patient’s family signed an informed consent prior to the treatment.A computed tomography (CT) scan revealed multiple high-density nodules associated with the left spermatic cord, which showed partial fusion, uniform density and a clear boundary with the testis . In addi+, Bcl-2+, partial CD34+, focal S-100+, SMA+ and CD68− , two common imaging examinations, play an important role in the diagnosis of SFTs. Ultrasound examination is more effective in the identification of solid and cystic tumors, while MRI is able to illustrate anatomical associations in the tumor surroundings more precisely . Isodensf an SFT . Positrorequired ,16. In t+, Bcl-2+ and CD99+, are generally positive in SFT cases, which has aided the diagnosis of SFTs (+ is indispensable in the diagnosis of an SFT; however, the expression of CD34 is not limited to SFTs (+, Bcl-2+ and CD99+ was demonstrated by immunohistochemistry examinations, confirming the diagnosis of an SFT.Previous studies have confirmed that tumor markers, including CD34 of SFTs ,18. Curr to SFTs . In the The conventional treatment for an SFT is the complete resection of the tumor with negative margins ,20 or reThe prognosis of SFTs is obscure for rare cases. Usually, an SFT is defined as a benign form of tumor due to its low rates or even absence of local recurrence and metastasis, as demonstrated by a number of previous studies ,2,22,23.In conclusion, pathological examination remains the standard approach for the diagnosis of SFTs, while radical resection of the tumor/s is the first choice for treatment. Furthermore, patients should undergo careful follow-up examination in order to identify tumor recurrence.


Pleomorphic adenomas are tumors mostly originating from salivary glands. These lesions in the larynx are very rare.We report a rare case of pleomorphic adenoma that originated from the mucosal lining, just above the glottic area at the level of the laryngeal ventricle in a 55-year-old female patient. The tumor could not be palpated easily but was observed in the CT scan. We resected the large and firm tumor using trans hyoid pharyngotomy as the surgical approach. Pleomorphic adenoma in the ventricle of the larynx is an extremely rare lesion. Trans-hyoid pharyngotomy can have good results as the surgical approach in removing such lesions. Most pleomorphic adenomas originate from the major salivary glands, especially the parotid gland. However, minor salivary glands may be the site of origin for some cases of this tumor. Such lesions in the larynx are very rare and reported cases in literature are less than 30. When the larynx is involved with this benign tumor, the most common site of origin will be the epiglottis (subglottis in some articles), but in our case, we describe a mixed tumor from the ventricle of the larynx that is not recorded in literature as the site of origin. Minor salivary glands may be the site of origin for pleormorphic adenoma in different areas such as the oral cavity, pharynx, retromulararea, nose, sinuses, parapharyngeal space, lacrimalgland, and even the breast -6.The total number of reported cases of laryngeal pleomorphic adenoma in literature are 23. In Cummings’ textbook of otolaryngology, the total reported cases are 27. In Boo-Ali-Sina medical center in Sari, Iran, another case of epiglottic mixed tumor is reported. Pleomorphic adenoma can originate in any part of the larynx, but some of the authors report the epiglottis and some of them report the subglottis as the most common site of this benign tumor . HoweverA 55-year-old female patient was brought to the emergency ward because of severe dyspnea and cyanosis and she went into the apnea state rapidly. During resuscitation and endotracheal intubation, an obstructing mass was seen in the laryngeal lumen and therefore she was intubated with great difficulty and then admitted to the ICU. She had dyspnea since 2 years, first in the exertional form and then even during rest. Six months before presenting with these symptoms she had a change of voice and dysphonia. When lying down on her right side, her dyspnea became more severe. She also complained of dysphagia but suffered no weight loss. She didn't smoke or drink alcohol and there was no history of infectious disease, or positive family history. During direct laryngoscopy and video laryngoscopy, a large and firm tumor was seen, filling about 90% of the intra laryngeal lumen. It was pink in color with a distinct and marked vascular pattern over its capsule .The pedicle of this mobile tumor was toward the lower parts in the larynx and could not be palpated easily. In the CT scan, the tumoral lesion was seen from the ventricle and above the base of the epiglottis .The excised tumor was an encapsulated tumor with a mucosal lining and its size was about 3.3 x 5.4 centimeters .Upon microscopic description, groups of epithelial cells in a mixoid ground were reported .Although the diagnosis of pleomorphic adenoma is easily possible with light microscopy in most cases, some special immunohistochemical stainings are also helpful . MitoticUpon review of literature, 27 cases of pleomorphic adenoma of the larynx were reported up to 2011. Like any other benign laryngeal tumor, symptoms of laryngeal mixed tumors include dyspnea, dysphonia or hoarseness, and dysphagia . RespiraCurrent approaches in use for the excision of laryngeal pleomorphic adenomas include lateral pharyngotomy, laryngofissure, and use of laser ,13. EpigCryotherapy was also used for treating the same lesions in the trachea. Possible complications in each of these approaches are recorded. Bilateral recurrent nerve paralysis was reported in a case treated with laser. In our patient, after transhyoid pharynogotomy a pharyngo cutaneous fistula was treated successfully with conservative procedures including aspirations and pressure dressings. Although complete surgical excision with a safe margin of normal tissue is the treatment of choice, the chance of recurrence cannot be ignored and the patient should be observed and followed in regular post op visits. In most cases, recurrence occurred 18 months after surgery . DespiteIn any case of hoarseness and/or shortness of breath, all parts of the larynx should be considered as the site of origin of a mass lesion. In benign tumors of the laryngeal ventricle, trans-hyoid pharyngotomy can be a good approach with acceptable results.


A 46-year-old female with a history of muscle weakness, nephrolithiasis and peptic ulcer. She was found to have hypercalcemia (115 mg/l), hypophosphoremia (16 mg/l) and increased PTH serum level (1090 pg/l). In order to discriminate between PH and pseudohyperparathyroidism, a solid-phase, two site chemiluminescent enzyme-labeled immunometric assay directed towards the intact PTH molecule was used and a diagnosis of primary hyperparathyroidism was made. Several studies were performed with the purpose to achieve a pre-operative localization of the oversecreting parathyroid tumor. A cervical sonography showed a normal thyroid gland and no evidence of the tumor was found in the neck. Therefore a 99-TC sesta MIBI was carried out and an accumulation area projected onto the anterior mediastinum was demonstrated (A). Finally a computed tomography scan of the thorax revealed a 4 cm nodule, in the thymic and paratracheal region of the anterior mediastinum (B). The patient underwent surgical resection of the tumor by a lower cervicotomy approach . Intraoperative parathyroid hormone measurements show a appropriate decrease in PTH levels. A 4x3x2 cm surgical specimen of a parathyroid tissu mass was removed. The anatomopathological examination confirmed the diagnosis of parathyroid adenoma. The post-operative course was uneventful and the patient was discharged in 4th post-operative day with an excellent functional and cosmetic result. A quarterly follow-up was started. PTH serum level and calcemia have been within normal ranges all through 18 months after surgery.


Small bowel tumours are rare and comprise less than 2 % of all primary gastrointestinal neoplasms. Among these tumours, a leiomyosarcoma belonging to soft tissue sarcomas is extremely rare and accounts for about 1 % of malignant mesenchymal lesions in the gastrointestinal tract. Due to its aggressive nature and slow growth, it is often diagnosed at the late stage when curative treatment is impossible. Authors report a first case of leiomyosarcoma with chronic recurrent ileocaecal intussusception and literature review to analyse diagnosis and treatment features of the ileum mesenchymal tumours.We present a case of an 87-year-old Caucasian man suffering from cramp-like abdominal pain for months. Due to lack of clinical signs and unspecific complaints, a diagnosis was delayed. Despite a detailed in-hospital examination, a proper diagnosis was established as late as during an operation. The patient was treated by surgery with good results. An uncommon laparoscopic resection of the small bowel with a tumour was performed. A histopathological investigation confirmed a very rare mesenchymal lesion of the distal ileum. The patient is under control with no recurrence for 1 year of the follow-up period.Reported case indicates that a usually asymptomatic tumour can cause uncommon chronic recurrent ileus signs. CT and MRI scans are investigation of choice in such cases, but they are sometimes inconclusive. It might be worth highlighting the good results of laparoscopic leiomyosarcoma lesion resection with a very good outcome. Abdominal pain is one of the most commonly reported ailments with no relation to the sex or age. It is often the first symptom of serious gastrointestinal tract diseases. Differentiation is difficult. Many of reported abdominal pain cases are caused by gastroenteritis or woman’s diseases. Sometimes, it can be caused by a very rare disease that shows unspecific signs and has to be well differentiated for curative treatment.We present a very rare case of a patient with severe abdominal pain in the right lower quadrant caused by intussusception of the small bowel with a leiomyosarcoma in the caecum with ileocaecal subocclusion.A leiomyosarcoma (LMS) is a rare smooth muscle malignant neoplasm which appears mostly in the retroperitoneal space, vascular wall  and soft tissues of the lower extremities. It is an exceptionally rare tumour of the gastrointestinal tract arising in such localisations like the oesophagus, stomach and small bowel. LMS belongs to a heterogeneous group of soft tissue neoplasms, and with its aggressive nature, it often presents local recurrence and visceral metastases. As in the presented case, clinical manifestation of this disease, as in other small bowel tumours, is unspecific. Authors report a very rare case of recurrent intussusceptions with tumour treated by laparoscopy with good results. To our best knowledge, there is only one published case of leiomyosarcoma treated by laparoscopic resection, but we have not found any papers concerning chronic recurrent intussusception caused by leiomyosarcoma.An 87-year-old Caucasian patient, suffering from arterial hypertension and hepatitis C, was admitted to the surgical department due to abdominal pain and weight loss of about 10 % during last 4 months.For 6 months, the patient had been complaining about cramp-like abdominal pain in the right lower quadrant and suprapubic area, vomiting and bloody stools 3–4 times last week. He also noticed that eating, but not drinking, had exacerbated pain which usually stopped without using any drugs within 3–4 h. His past medical history revealed a stomach resection due to a peptic ulcer with relaparotomy due to bleeding immediately after an operation and cholecystectomy about 20 years ago.On admission, the patient was in a good condition, with adequate logical responses and without complaints. No palpable mass in the abdomen was noted during an examination. Laboratory test revealed WBC 6250/uL, RBC 4,150,000/uL, PLT 263,000/uL, and low-grade anaemia Hb 11.8 g/dL. The cancer antigen levels were normal: carcinoembryonal antigen (CEA) 2.29 ng/mL  and CA 19-9 antigen 16.36 IU/mL . A computer tomography (CT) scan confirmed the presence of two suspected findings in the sixth and eighth liver segments, deep in the stroma. Results remained unclear, and magnetic resonance imaging (MRI) was performed with no conclusion. Neither CT scan nor MRI did not reveal any evidence of tumour and intussusception probably due to lack of occlusion signs at the moment. A colonoscopy performed revealed a caecal tumour, probably involving Bauhin’s valve , the patient was qualified for a laparoscopy in order to perform right hemicolectomy. During a surgery, intussusception of about 50 cm of the non-gangrenous small bowel into the caecum was found Fig. . Liver iPostoperative histopathological results revealed a tumour of 5 × 4 × 3.7 cm, C-kit (−), CD34 (−), S100 (−), actin (+), high mitotic activity—28 figures per 10 HPFs, and high degree of necrosis Fig. .Fig. 4HiThe postoperative period was uneventful; no complications were noticed. The patient, after intensive postoperative rehabilitation, was discharged on day 7, in a good condition.After a consultation with an oncologist, patient was disqualified from adjuvant therapy. Due to hepatitis C, advanced age and past surgical history, he was also not qualified for a liver metastases resection.The 12-month follow-up period completed, and no recurrence signs were noticed. Liver metastases are stable in CT imaging tests.Small bowel tumours (SBT) are rare and comprise less than 2 % of all primary gastrointestinal (GI) neoplasms , 2. Therhttp://www.cap.org/apps/docs/committees/cancer/cancer_protocols/2013/GIST_13protocol_3022.pdf). DOG1, chloride channel protein named “Discovered-On-GIST 1”, was published as a useful tool in GIST differentiation but was not listed in our local protocols in the time of diagnosis [http://www.cap.org/ShowProperty?nodePath=/UCMCon/Contribution%20Folders/WebContent/pdf/cp-gist-14protocol.pdf).LMS is a rare smooth muscle malignant neoplasm and belongs to soft tissue sarcomas (STS). In late 1990s, due to the introduction of new immunoassay tests, there were changes in the classification of GI mesenchymal tumours, and currently, most of them are classified as gastrointestinal stromal tumours (GIST). Primary leiomyosarcomas are diagnosed rarely. These are aggressive neoplasms often with severe cellular atypia and numerous mitoses. Definitive tools allowing for differentiation leiomyosarcoma from GIST are actin and desmin positive reactivity, CD117, CD34 and PDGFRA  negativity. According to 2013 Protocol for the Examination of Specimens from Patients With Gastrointestinal Stromal Tumor (GIST) that we based our diagnosis on, stainings listed above were done in the specimen obtained from our patient   as a nuclear antigen-2 protein and specific EBV receptor in smooth muscle cells are expressed in immunocompromised individuals .Generally, STS prognosis is poor; the median overall survival is about 12 months . A 5-yeaSeveral histologic grading systems have been validated in soft tissue sarcomas, including LMSs; but no system is currently accepted worldwide. According to the World Health Organisation (WHO), the grading of sarcomas should be performed using the US National Institute of Health (NIH) or French Federation Nationale des Centres de Lutte Contre le Cancer (FNCLCC) grading systems. The ‘French system’ evaluates three tumour parameters: a degree of tumour differentiation, number of mitotic figures per 10 HPFs and a degree of tumour necrosis. The American system uses a histological diagnosis coupled with a degree of cellularity, cellular polymorphism and mitotic activity. A degree of necrosis is also identified, and a result greater than 15 % diminishes the prognosis. It has been demonstrated that the FNCLCC results show a better correlation with overall and metastasis-free survival , 14.Due to late clinical manifestation, tumours are diagnosed rather late when subileal or GI perforation has already developed. Permanent bleeding into the gastrointestinal tract has to be confirmed by a faecal occult blood test as there is usually no massive haemorrhage. Nausea, vomiting, flatulence and abdominal pain are often devaluated and delay a diagnosis .In the presented case, the main complaint was recurrent abdominal pain diagnosed by a general practitioner (GP) as gastroenteritis and treated with a muscle relaxant and stomach protective drugs. There were no signs of a subileal or abdominal mass on palpation; therefore, a GP decided to send the patient for further tests when no recovery was observed. Intussusception was not suspected.It is worth mentioning that intussusception was described for the first time in 1674, and successful operation in a child was performed in 1871 . This diWith regard to our imaging tests, both CT and MRI scans did not confirm any diagnosis, and the only findings included suspected metastases in the liver. It is worth highlighting that, due to clinical signs before admission, probably, intussusception and suboclusion occured and reduced spontaneously many times what caused investigation difficulties. A colonoscopy confirmed the presence of a tumour, but wrong localisation on Bauhin’s valve was described. Histopathological findings from colonoscopy samples were also not diagnostic with regard to a type of a neoplasm. The patient was qualified for a laparoscopy in order to perform right hemicolectomy as a caecal tumour was suspected in a preoperative examination. During a surgery, when intussusception of the ileum into the caecum was diagnosed and a small bowel tumour was confirmed, we had to change an operating procedure into an ileal resection.Laparoscopy procedures in case of smooth muscle neoplasms are rare. As a result of searching the PubMed database, only one case of a segmental laparoscopic resection of the small bowel due to a leiomyosarcoma described by Hamm et al. in 2013 was found . These nWhen compared to the results of other reports concerning colon cancer treatment, our report demonstrates that a laparoscopic resection of small bowel sarcomas may be a reasonable and safe alternative to a usually performed open resection.As it was already mentioned with regard to current histopathological classification, smooth muscle neoplasms are very rare—gastrointestinal stromal tumours are predominantly diagnosed. Proper differentiation between these two types of STSs is very important to select appropriate adjuvant therapy. In some cases, leiomyosarcomas can be treated by conventional chemotherapy, whereas GIST therapy requires different drugs such as imatinib—a tyrosine kinase inhibitor , 26. In Chronic recurrent cramp-like abdominal pain can be caused by serious gastrointestinal disease and often requires endoscopic investigation, especially in elderly patients. Intussusception in these cases might not be permanent; it occurs and reduces spontaneously but recurs. This can cause diagnostic problems and delays final diagnosis. A surgical resection, also by laparoscopy, is the treatment of choice and allows a patient to benefit from adjuvant therapy.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of written consent is available for review by the Editor of the journal.The authors confirm that the content of the manuscript has not been published, or submitted for publication elsewhere.This case report is a retrospective study to report some important data concerning investigation and treatment of the patient. Informed consent was obtained. No ethics is required for study.


Pulmonary hamartomas are more common than expected because they are usually asymptomatic and are either discovered on routine chest radiography or when they are noted incidentally in approximately 0.25 % of autopsies. In contrast, pulmonary fibroleiomyomatous hamartoma, which consists of interlacing bundles of smooth muscle cells admixed with fibrous tissue and numerous tubular or cleft-like epithelial inclusions, is a rare type of hamartoma. Controversy exists regarding the pathogenesis of this tumor. We herein present a rare case of a 68-year-old male patient without a pre-existing smooth muscle tumor, who underwent resection for a tumor that was considered to be a true pulmonary fibroleiomyomatous hamartoma. Hamartomas are a relatively common form of benign lung tumor. Fibroleiomyomatous hamartomas, on the other hand, are rare. Pulmonary fibroleiomyomatous hamartoma was first described as “diffuse fibroleiomyomatous hamartomatosis” by Cruickshank et al. in 1953 , 3. ThesThe patient was a 68-year-old asymptomatic male who visited our hospital after an abnormal shadow was suspected on a chest X-ray. He had undergone surgery for colon cancer 8 years previously. He had no history of smooth muscle tumors nor did he have any history of occupational exposure to silica, beryllium, or asbestos. Chest computed tomography (CT) revealed the presence of a well-demarcated solid nodule in the right upper lobe adjacent to the pleura, which measured 1.0 cm in diameter . Three ports were placed in the left lateral decubitus position. The lung was deflated to confirm the right lung nodule. A linear stapling device was used to resect the lung nodule. The nodule was firm, solid, yellow-white in color on sectioning Fig. , and wasSingle or numerous focal proliferations of the smooth muscle tissuePulmonary lymphangioleiomyomatosisTuberous sclerosis with pulmonary changesApart from typical hamartomas, Spencer  identifiAccording to Spencer, very rare focal changes occur in the walls of the pulmonary alveoli, in the pleural region, and in the walls of the small bronchi. In our case, the bronchial epithelium extended into the alveolar septa in the pulmonary parenchyma without a cartilaginous or adipose element. Spencer suggested that they mainly developed subpleurally and extended towards the hilum. In our case, they reached the subpleural space. Patients with the lesions are generally asymptomatic. Multiple lesions occur more frequently than solitary lesions, and the tumors are more prevalent in women. There are many reports on uterine leiomyomas metastasizing to the lungs .fibroleiomyomatous hamartoma or benign metastasizing leiomyoma could include some cases of low-grade leiomyosarcomas, true benign metastasizing leiomyoma, and true fibroleiomyoma [fibroleiomyomatous hamartoma or benign metastasizing leiomyoma should be reconsidered to avoid misleading diagnoses. In the present cases, the lesion was solitary and occurred in a male who had no history of smooth muscle neoplasm. Thus, true fibroleiomyomatous hamartoma was considered to be the most suitable the diagnostic term to describe the patient’s condition.The histological characteristics of what is currently referred to as fibroleiomyomatous hamartoma or benign metastasizing leiomyoma are as follows: (i) a well-circumscribed nodule composed of smooth muscle cells with numerous epithelial inclusions and (ii) no cellular atypia or mitotic figures , 7. In oeiomyoma . Since cFour partial resections and one lobectomy are reported to have been performed in the five reported cases of pulmonary fibroleiomyomatous hamartoma. There are no reports of the postoperative recurrence of pulmonary fibroleiomyomatous hamartoma , 3. We tWe herein presented a rare resected case of a solitary pulmonary fibroleiomyomatous hamartoma and discussed its differential diagnosis and histogenesis.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.CT, computed tomography; SMA, smooth muscle actin; VATS, video-assisted thoracoscopic surgery