Spectral-domain optical coherence tomography findings in a patient with acute macular neuroretinopathy, and correlation with functional defects on microperimetry, are presented.A 25-year old Caucasian woman presented with bitemporal field defects following an upper respiratory tract infection. Her visual acuity was 20/20 in both eyes and a dilated fundus examination revealed bilateral hyperpigmentary changes in the papillomacular bundle. Our patient underwent further evaluation with spectral-domain optical coherence tomography, infrared and fundus autofluorescence imaging. Functional changes were assessed by microperimetry. Infrared imaging showed the classic wedge-shaped defects and spectral-domain optical coherence tomography exhibited changes at the inner segment-outer segment junction, with a thickened outer plexiform layer overlying these areas. Fluorescein and indocyanine green angiography did not demonstrate any perfusion defects or any other abnormality. Microperimetry demonstrated focal elevation in threshold correlating with the wedge-shaped defects in both eyes.Spectral-domain optical coherence tomography findings provide new evidence of the involvement of the outer plexiform layer of the retina in acute macular neuroretinopathy. Acute macular neuroretinopathy (AMNR) is a rare condition characterized by wedge-shaped lesions pointing towards the foveal center, resulting in bilateral or unilateral scotomas, typically with preserved central visual acuities ,2. The aWe present findings of infrared (IR) imaging and spectral-domain OCT  and correlate these with retinal function by microperimetry. The findings demonstrate outer plexiform layer (OPL) thickening in this case of AMNR.® Migraine  tablets. She used Midrin  as needed for her headaches concurrently. Additionally, she smoked half-pack cigarettes and consumed four to five 12-ounce cans of a caffeinated drink, Mountain Dew (caffeine 54 mg/can) per day.A 25 year-old Caucasian woman presented with a four-day history of acute onset of blurred vision in both eyes. She reported a viral upper respiratory tract infection for seven to 10 days, for which she had taken two ExcedrinHer uncorrected Snellen visual acuity was 20/20 in both eyes and Amsler testing revealed bitemporal paracentral scotomas. She correctly identified 10 and nine out of 14 Ishihara color plates, in her right and left eye, respectively. No afferent pupillary defect was noted and the anterior segment was unremarkable. Fundus examination revealed bilateral hyperpigmentary changes in the papillomacular bundle Figure . Fundus Five months after initial presentation, her color vision improved to 14 of 14 Ishihara color plates correctly identified in each eye. Repeat HVF testing demonstrated interval improvement in the scotomas, more in her right eye than left. Similarly, SD-OCT showed a corresponding small improvement at the IS-OS junction in her right eye Figure  and 2B aAMNR remains an elusive condition in regards to the etiology of retinal lesions. Eighty-three percent of cases affect younger women, nearly half of whom report an associated viral illness . Other rIn our patient, the characteristic lesions were not seen on fundus examination, but were clearly evident on IR imaging. Fundus autofluorescence did not demonstrate an abnormal autofluorescence pattern, indicating that the retinal pigment epithelium was not affected. Two recent reports demonstrated localization of the retinal lesions in AMNR to the photoreceptor IS/OS junction, using ultra-high resolution OCT ,6. SD-OCWritten informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.VB and SG were responsible for the clinical follow-up of our patient. RM, SG and KC were responsible for editing and critical review of the manuscript. All authors have read and approved the final manuscript.


Torsion of the gallbladder is an extremely rare cause of acute abdomen, which commonly affects thin elderly women. A prompt surgical approach is necessary to avoid fatal complications associated with gangrene and perforation of the gallbladder. However, it is difficult to make a preoperative diagnosis using ordinary imaging modalities.An 84-year-old Japanese woman was admitted to our hospital due to left lower abdominal pain. Her pain shifted suddenly to the right upper abdomen a half day after admission. Although her enlarged and wall-thickened gallbladder had been already seen at admission, it rotated approximately 180 degrees and deviated to the midline of her abdomen on the second computed tomography scan, which helped us to make a correct diagnosis of gallbladder torsion. The patient underwent an emergency operation (detorsion and cholecystectomy) and recovered without any complications. The gallbladder had necrosis due to torsion.Sequential diagnostic imaging might be helpful to make a preoperative diagnosis of gallbladder torsion when the gallbladder is enlarged and wall thickened but the patient does not present with typical clinical symptoms. Torsion of the gallbladder is an extremely rare condition that was first reported in 1898 by Wendel ,3. CliniWe report here a case of torsion of the gallbladder in an elderly woman without typical clinical symptoms, in which sequential computed tomography (CT) scans helped us to make a correct diagnosis preoperatively.2 and her vital signs were all within normal limits. Physical examination demonstrated tenderness in the left lower abdomen without Blumberg’s sign and with no demonstration of Murphy’s sign. Laboratory data were all within normal limits including inflammatory response, liver function tests, and biliary enzymes. A CT scan of her abdomen at admission demonstrated an enlarged and wall-thickened gallbladder without gallstone, prominent small bowel gas, and mild intestinal dilatation and wall thickening (Figure An 84-year-old woman was admitted to our hospital with complaints of left lower abdominal pain and nausea without fever. Her past surgical history included a hysterectomy and a femoral neck fracture repair. She also suffered from hypertension and hyperlipidemia. Her body mass index was 18.2kg/mThere are two types of gallbladder torsion : one is The decisive findings of gallbladder torsion are described in the literature as left-sided enlarged gallbladder, thickened gallbladder wall without enhancement effect, and a cystic duct located on the right side of the gallbladder ,8. HowevAs for the treatment for torsion of the gallbladder, prompt detorsion and cholecystectomy are required to avoid potentially fatal sequelae of gangrene and perforation of the gallbladder . We chosTorsion of the gallbladder is a rare cause of acute abdomen and it is difficult to diagnose preoperatively. It is important to take this disease into consideration in the differential diagnosis of acute abdomen in all elderly patients. Moreover, sequential diagnostic imaging including CT scans might be helpful when the gallbladder is enlarged and wall thickened but the patient does not present with typical clinical symptoms.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.TK and KS diagnosed, investigated and managed the patient. Also, TK and KS determined the medical significance and wrote the manuscript. All authors read and approved the final manuscript.


We present an uncommon case of traumatic pseudolipoma in a 24-year-old female, causing facial asymmetry. Literature review suggests trauma as a possible etiology for its pathogenesis, which was present in this case. Microscopically, sometimes it is difficult to differentiate between normal adipose tissue and lipoma. Clinician must provide accurate clinical information in order to make a definitive diagnosis of traumatic pseudolipoma. Its pathogenesis has also been highlighted in this article. Lipomas are benign mesenchymal neoplasms composed of mature adipocytes, usually surrounded by a thin fibrous capsule. They areThe term traumatic pseudolipoma was proposed by Brooke and MacGregor. Some speculate, trauma serves as a cause of fat necrosis, may trigger the formation of lipoma “like”, so called “traumatic pseudolipoma.” Several reports of similar cases, all subsequent to trauma, gave evidence for the acceptance of the term traumatic pseudolipoma.–10A 24-year-old female reported with a chief complaint of asymptomatic swelling of the left side of the face, since 4 years. She gave history of trauma 4 years back in the same region, due to pressure cooker burst. Since then, the swelling had reduced to the present size and is constant since 1 year.Extraoral examination revealed a swelling, measuring 3 × 3 cm in diameter, extending superiorly from inferior orbital process, mesially up to ala of the nose, distally to zygomatic process and approximately 3 cm inferiorly . On palpAs the mass was deep seated , fine needle aspiration (FNA) cytology was done. Aspiration consisted of jelly-like colorless material. Slides were prepared, air dried and stained with Giemsa stain. On cytopathological examination, the smear showed few areas of cellular material composed of vacuolated cell. Few of these cells showed nucleus at the periphery, which was suggestive of mature adipocytes .Surgical excision with facial recountering was done under general anesthesia via naso-endotracheal intubation. Microscopic examination of the excised soft tissue mass revealed sheets of mature adipocytes containing large clear cytoplasm with eccentric nuclei. There was no evidence of cellular atypia or metaplasia . The tumPseudolipoma of the buccal fat pad into the oral cavity was first reported in literature by Clawson in 1968. It was fThis disease event typically occurs in children ranging from 4 months to 4 years of age when the fat pad is still quite large in proportion to the size of the face. The etiology of traumatic pseudolipoma of the buccal fat pad is speculated in infants and young children because of their frequent chances of falling down with an instrument such as a toothbrush in their mouth, and getting an oral cavity injury. Furthermore, negative intraoral pressure induced by their suckling activity may promote herniation of the buccal fat pad from the wound.–1013141013The origin of these tumors, whether hamartoma or neoplasm is subject to question. Brooke aet al. and the development of post-traumatic lipomas.[Recently, a genetic defect with translocation or partial loss of chromosome 12 has also been suggested as a factor contributing to the formation of lipomas. In addition, chronic local compression causing anatomical defects in the fascia may also result in pseudolipoma formation. Herbert and DeGeus reported a patient with an abdominal lipoma induced by wearing tight trousers. An extensive study on the origin of post-traumatic lipomas postulated that subcutaneous scar formation and contracture after a soft tissue trauma may also lead to the development of pseudolipomas. Recently lipomas. In the pde novo lipoma, and is simply described as normal fat in an abnormal location. Cells of the lipoma differ metabolically from normal fat cells. Furthermore, Fatty acid precursors are incoporated at a more rapid rate into the lipoma than into normal fat while lipoprotein lipase activity is reduced.[Histologically, it is indistinguishable from normal fat or  reduced.We present a case of subcutaneous fibrofatty lesion. It presented as a swelling in the cheek region causing facial disfigurement, as a result of trauma. Though aspiration cytology has little value in the diagnosis of superficial lipoma, it may be helpful in elucidating the nature of deep-seated tumor or tumor in unusual location. Clinical information provided by the clinician and its histopathological features helped in making a definitive diagnosis of “traumatic pseudolipoma”. Further clinical and experimental studies are necessary to clarify the pathogenesis and the mechanism of post-traumatic lipoma formation.


Tenosynovial giant cell tumor is a locally aggressive tumor arising from the synovia of the fibrous tissue surrounding the joints, tendon sheaths, mucosal bursas, and tendons. Although it is often to be observed at the hand, localized form is very rare in the knee joint. In this case report, we aimed to present a very rare case of a surgically treated intra-articular giant cell tenosynovial tumor arising from the hoffa's infrapatellar fat pad of a 19-year-old male patient, by reviewing the literature. The patient we have treated with marginal excision was asymptomatic at the 14th month in the controls and recurrence was not detected. Tenosynovial giant cell tumor is a locally aggressive tumor arising from the synovia of the fibrous tissue surrounding the joints, tendon sheaths, mucosal bursas, and tendons –3. Tenos19-year-old male patient was admitted to our clinic with complaints of swelling and pain increasing with activity in his left knee. The patient had no pain at rest. On the examination of the patient there was a 2 × 3 cm in size swelling that appeared during flexion at the lateral side of the infrapatellar region of the left knee. Lachman, anterior drawer, posterior drawer, and medial and lateral stress tests were negative. Mc Murrey and Apley tests were also negative. The laboratory tests were normal. No abnormal findings were found at the X-ray. A heterogeneous, 3 × 2 cm sized, well-circumscribed lesion localized into the infrapatellar fat pad in the left knee was present at the MR-imaging. The lesion was hypointense on T1-weighted images and hyperintense on T2-weighted images (Figures Operation was planned because the lesion was symptomatic. Although the lesion was well-circumscribed, the lesion was heterogeneous, pathologies such as synovial sarcoma could be observed in this region, so, firstly, ultrasonography-guided needle aspiration biopsy was performed. Cytopathologically malignant cells were not seen and giant cell tumor of tendon sheath was reported. Thereupon marginal excision with open surgery was decided.Three cm longitudinal incision was made at the lateral side of patellar tendon of the left knee in the supine position under spinal anesthesia under tourniquet. After reaching the joint capsule, the lesion was reached by longitudinal arthrotomy. The lesion was excised marginally. Giant cell tenosynovial tumor was diagnosed at the histopathological examination .Postoperative range of motion exercises were started the day after surgery. The patient was asymptomatic at the 14th month control and recurrence was not detected.Tenosynovial giant cell tumors originate from the synovial tissue of various regions of the body –8. It isMarginal excision is sufficient in the treatment. Recurrence rate is between 10 and 20% . In the As a result although tenosynovial giant cell tumors localized in the knee joint are very rare; they should be kept in mind in the differential diagnosis. MRI is helpful in diagnosis and marginal excision is sufficient in the treatment.


Extramammary Paget’s disease is an uncommon intraepithelial neoplasm that arises in areas rich in apocrine glands. Treatment includes wide surgical excision and nonsurgical modalities. We present the case of a patient with perianal Paget’s disease with no recurrent disease after wide surgical resection.Our patient was a 46-year-old man of Macedonian ethnicity who presented with a pruritic perianal lesion measuring up to 6cm without pain or bleeding. Two biopsies and a perianal wide surgical excision were performed. The tissue specimens were formalin-fixed and the paraffin-embedded samples analyzed according to standard histochemical and immunohistochemical procedures.Surgical perianal skin excision revealed diffuse eczematoid, whitish plaques. Pathohistology showed Paget cells infiltrating his epidermis and adnexal epithelium, with ulceration. Immunohistochemical analysis revealed positive Paget cell expression for cytokeratin 7, epithelial membrane antigen, carcinoembryonic antigen, androgen receptor and human epidermal growth factor receptor 2, and negative expression for cytokeratin 20 and melan-A.Paget’s disease is a rare disorder that should be considered in the differential diagnosis of perianal lesions. Reporting cases of extramammary Paget’s disease is crucial for diagnostic guidelines and different therapeutic options. Extramammary Paget’s disease (EMPD) is a rare cutaneous neoplasm that mainly affects the elderly. It predominantly involves apocrine gland-bearing areas, especially the vulva, scrotum and perianal region, but also the axilla, groin, thigh, external ear, umbilicus and nose . PerianaA 46-year-old man of Macedonian ethnicity presented with a pruritic perianal lesion measuring up to 2cm without pain or bleeding. Our patient was diabetic and had a family history of diabetes and hypertension. Five months after his initial presentation, a colonoscopy and a biopsy were performed at Clinical Hospital Sistina - Adzibadem. The next month, our patient was admitted to our University Clinic of Digestive Surgery, where a physical examination revealed a perianal eczematous lesion measuring 6 × 4cm and enlarged inguinal lymph nodes. A second biopsy with a left lymphadenectomy was performed.The tissue specimens were formalin-fixed and paraffin-embedded at our Institute of Pathology. We used a routine hematoxylin-eosin stain and performed additional histochemical and immunohistochemical analysis, including staining with Alcian blue and for cytokeratin (CK)7, CK20, epithelial membrane antigen, carcinoembryonic antigen, melan-A, androgen receptor and human epidermal growth factor receptor 2 (Her2/neu).Microscopic analysis of the biopsy specimens showed large Paget cells with abundant pale cytoplasm, and large nuclei infiltrating the basal part and the whole thickness of the squamous epithelium and adnexal epithelium. Occasional cells had a signet-ring appearance. His inguinal lymph node revealed reactive lymphadenopathy. A diagnosis of EMPD was made.in situ PPD was made.The perianal surgical skin excision measured 5.5 × 6.5 × 0.7cm and showed diffuse ulcerated eczematous plaques Figure . HistopaAfter two biopsy findings of EMPD, a wide surgical excision was performed Figures  and 9. Tin situ epithelial form without associated carcinoma, an epithelial form with associated adnexal carcinoma, and association with visceral malignancy. Paget cells have the potential to invade the dermis and to metastasize. The anatomic site of the presenting EMPD is strongly related to the underlying visceral carcinoma in 86% of cases [EMPD is a rare cutaneous intraepithelial adenocarcinoma arising in apocrine gland-bearing areas, particularly the perineum, vulva, axilla, scrotum, penis and groin. PPD mostly refers to intraepithelial carcinoma within 6cm of the anus and below the dentate line . Patternof cases . Our patClinical presentation is often nonspecific and the diagnosis is frequently overlooked. Patients commonly have symptoms like pruritus, irritation and rash, although pain and bleeding may occur in long-standing cases. These lesions are typically erythematous, or whitish gray, dry and raised, but may turn into eczematoid, ulcerated, nodular or papillary forms -10. It iDifferential diagnoses include Bowen’s disease, contact dermatitis, lichenoid lesions, psoriasis, melanoma, perianal Crohn’s involvement, mycosis fungoides, squamous cell carcinoma and tinea cruris.et al.[Histopathology reveals large, round, clear-staining cells with abundant pale cytoplasm confined to the epidermis. The nuclei are large and situated in the periphery of cells. In the lower epidermal layers, glandular clusters can be seen that are absent in mammary Paget’s disease. Liu et al. support et al..Pathogenesis of PPD is controversial. Helwig and Graham  considerA patient diagnosed with EMPD needs an initial clinical assessment, evaluation of the extent of involvement of the lesion, and work-up for a possible underlying malignancy. A wide range of treatment modalities have been reported, including surgical and nonsurgical approaches. Surgical methods are still the mainstay in the management of PPD, including wide local excision with or without reconstruction and grafting, abdominoperineal resection, and Mohs micrographic surgery. Radiotherapy, chemo-radiotherapy and photodynamic therapy have been employed ,7,13. Dein situ PPD is favorable.Prognosis depends on whether the disease extends beyond the epidermis and the adnexal epithelium. If it is associated with subjacent adnexal carcinoma or regional visceral carcinoma, the prognosis is poor. Survival of patients with Long-term follow-up of patients with EMPD is required to exclude the recurrence of the disease and development of an associated cancer. Follow-up should include a punch biopsy from the margin of the old perianal lesion once a year, in addition to a colonoscopy once every two years .We present the case of a patient with PPD treated with wide surgical excision. Our patient has demonstrated a disease-free period of six months. The diagnosis was made using pathohistology and immunohistochemistry. Although surgical excision is the proposed treatment of choice, other therapy modalities are used but not yet standardized. Long-term follow-up is essential to detect local recurrence and development of invasive Paget’s disease or anorectal carcinoma.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.CK: cytokeratin; EMPD: extramammary Paget’s disease; PPD: perianal Paget’s disease.The authors declare that they have no competing interests.NJ performed the biopsies and wide surgical excision, analyzed the data and followed-up the patient. LS, VJ and BD performed the histological examination of the specimens and interpreted the patient data. NJ and BD were major contributors in writing the manuscript. All authors have read and approved the final manuscript.


Introduction. Each year around 2,200 people in the UK are diagnosed with laryngeal SCC . Compared to pharyngeal carcinoma, it is a highly curable disease with a survival rate of around 60% for all stages and all forms of treatment. Case Presentation. We present the case of a 60-year-old man with a previously treated T4 N2c transglottic squamous cell carcinoma (SCC), who developed an isolated swelling in the extensor compartment of his right forearm at 6 months after radical laryngectomy with bilateral neck dissection. Fine needle aspiration of the forearm lesion revealed SCC consistent with a metastasis from the laryngeal primary. MRI revealed that the lesion was confined to the muscle. Initial staging CT showed no distant metastases or signs of advanced disease, including no evidence of axillary nodal involvement. Conclusion. This case is therefore unusual, as one of only 2 cases reported in the scientific literature of isolated distant muscular metastasis from a laryngeal squamous cell carcinoma. We conclude that any muscular swelling, in the setting of previous head and neck malignancy, should be treated with a high degree of suspicion for metastasis and investigated promptly. Each year around 2,200 people in the UK are diagnosed with laryngeal SCC . CompareA 60-year-old male presented to the ENT clinic in November 2008 with a two-month history of dysphonia. There were no other symptoms apart from lethargy. His past medical history included mild chronic obstructive airway disease which required no treatment. He was a cigarette smoker: 40 pack years and consumed approximately 20 units of alcohol per week mostly in the form of spirits.On examination, he appeared systemically well, with no peripheral stigmata of chronic disease. Examination of the neck revealed bilateral cervical lymphadenopathy. There were multiple small (<2 cm), firm, well-circumscribed masses bilaterally which were mostly distributed within levels II-III of the neck. Flexible nasendoscopy performed in the clinic showed a fixed left hemilarynx. Examination under anaesthesia confirmed the findings of the flexible nasendoscopy, a large tumour of the left vocal cord crossing the anterior commissure to the right side along with slight subglottic extension.A staging CT scan of the head and neck was undertaken which showed a transglottic mass with significant local invasion and confirmed the bilateral enlarged cervical lymph nodes. No distant metastases were noted, including clear lung fields . Two weeks after presentation, total laryngectomy and bilateral modified radical neck dissections were performed. The histology revealed a 3.5 cm moderately differentiated (grade 2) SCC with microvascular, perineural, and thyroid cartilage invasion (T4). In addition, bilateral lymph nodes were positive for metastatic SCC with extracapsular spread evident on the left side (<6 cm greatest dimension). This cancer was therefore staged as T4N2c.In January 2009 (2 months postoperatively) the patient attended a routine follow-up appointment. He was well and had no symptoms of local recurrence or distant metastatic spread; however, on examination a small parastomal lesion was seen. Prompt local excision of this was performed and histology showed a parastomal recurrence. As a consequence, chemoradiotherapy was started. Six months after the initial presentation, the patient returned with a 5-week history of a mild swelling in the extensor compartment of his right forearm (May 2009). The forearm was swollen and mildly tender, but there was no erythema, and nothing sinister was suspected . A shortDifferential diagnoses regarding isolated limb swellings are plentiful and diverse and include cellulitis, trauma, postsurgical lymphoedema, DVT, and haematomas. These are all likely to be considered before malignancy. 72% of cases of laryngeal carcinoma occur over the age of 60 with the peak between 75 and 84 . In addi15–60% of our cardiac output supplies the skeletal muscles at rest and during exercise, respectively. If the body mass index is within the normal range, around 50% of the body is muscle. The musculature forms a significant proportion of our body and is well perfused; yet muscular metastases are uncommon.Malignancies usually follow a step-wise pattern of progression, starting with local invasion, followed by lymphatic then haematogenous spread. With laryngeal cancer, local invasion of tissues often gives rise to the common presenting symptom of hoarseness (particularly with glottic tumours). Later, this may result in fixation of the vocal cords. Considering lymphatic drainage, the nodes of the internal jugular chain are most commonly involved in laryngeal cancer . LymphatSuccessful distant metastasis relies on a series of steps: tumour transformation, dissociation and local invasion, intravasation, metastatic site stasis and extravasation, proliferation, and sustained growth. At each step there are various obstacles to overcome. Although the lymphatic and haematogenous systems are said to be in communication to an extent, survival of metastatic cancer cells in the blood stream appears far more difficult. In the 1970s, Fidler used radiolabelled cells to show that 99% of infused tumour cells are cleared from the blood within 24 hours . This waThis case illustrates why any distant swelling in a head and neck cancer patient should be treated with a high index of suspicion with regards to metastasis. Despite their initial innocuous appearance, imaging and histological investigations should be performed promptly to avoid any delay in diagnosis and treatment. Although muscular metastases are rare, they represent a very poor prognostic indicator judging by the life expectancy of these patients in the literature .


We are presenting the case of a 44–year old patient with large, well–differentiated liposarcoma of the right thigh. We are discussing the clinical findings, diagnosis and surgical treatment. The large dimensions (27/25 cm) and the origin of the tumor in popliteal fossa, migrating through the adductor canal  in the anterolateral muscular space of the right thigh, represent the particularity of this case.  Lipomatous tumors are the most common soft tissue tumors . Over tiA 44 year–old male patient was admitted in our clinic with a giant tumor in the right thigh, which started to grow two years before, with progressive increase in size. The local examination of the right thigh revealed a 35/30 cm high consistency tumor, with reduced mobility. There was no right inguinal adenopathy. Blood laboratory tests showed no changes from the normal range. Radiographic examination of the right thigh showed inhomogeneous enlargement of soft parts at this level, with linear opacities. The bone segments did not reveal pathological changes. Ultrasound evaluation showed the formation of a very poorly vascularized giant heterogeneous ultrastructure, which altered the vascular structures of the thigh muscle. A CT scan of the right thigh revealed the presence of an expansive process located on the anterior aspect of the thigh starting from the groin to the suprapatellar level, bordered above by sartorius muscle and right femoral muscle and posterior by iliopsoas muscle. Expansive processes measured approximately 11/19 cm (in cross sections), showing that the tumor was well defined, predominantly fatty in density and with numerous irregular septa inside, showing minimal contrast. The femoral artery and vein along with the popliteal vessels showed normal permeability. There were no changes in bone cortex of the right femur and no presence of inguinal lymph nodes. During surgery, a large, well–marked encapsulated tumor , was found, situated between the adductor muscles and the large muscles that push muscles aside. A simple tumor excision was carried out. The pathology exam showed neoplastic proliferation with predominant fat in the form of islands separated by thick well–vascularized conjunctive septa, with areas of tumor necrosis. Adipocytes had nuclei with increased volume and with low pleomorphism. Final diagnosis was well–differentiated ‘lipoma–like’ liposarcoma. The patient's state was favorable after the operation, without signs of local or systemic relapse in subsequent follow–up during the next 5 years. The patient did not receive adjuvant radiotherapy or chemotherapy.Liposarcomas represent 10–35% of all soft tissue sarcomas, making them the second most common type after malignant fibrous tumors. The WHO classification of soft tissue tumors (2002) divided liposarcomas into five distinct histological subtypes: well differentiated, dedifferentiated, mixoid, pleomorphic and mixed. The diversity of these lesions is reflected by their clinical and biological behavior, which ranges from non–metastatic tumors (well–differentiated liposarcoma) to tumors with high metastatic potential (pleomorphic liposarcoma) . BecauseSupernumerary circular and giant rod chromosomes genetically characterize all subtypes of well–differentiated liposarcomas. These chromosomal abnormalities contain amplification of the 12q13–15 region, which amongst other genes includes MDM2 and CDK4 , 9. MaliThe image on CT or MRI exams and the morphological relationship between the areas of fat and non-fat components allows the identification of the histological subtype of liposarcomas. Thick connective septa (>2mm) inside of a poorly vascularized fat tumor are suggestive of the diagnosis of well–differentiated liposarcoma , 13. TheWell–differentiated liposarcoma most frequently affects the deep soft tissues of extremities (65–75% of cases); over 50% of these are located in the lower limbs, especially the thigh . MacroscThe peculiarity of this case is represented by the large size and the origin of the tumor in popliteal fossa, migrating through the adductor canal  in the anterolateral muscular space of the right thigh. Liposarcoma is one of the most common forms of soft tissue sarcoma, presenting a broad spectrum of clinical behavior. This is closely related to the histological type of liposarcoma and patient management, the evolution being different for each case. Liposarcomas are usually well–differentiated tumors with non-metastatic potential, especially if they are located in the extremities. Understanding and recognizing the broad spectrum of radiological aspects and pathological bases of extremities, liposarcomas allow an improved management of these patients. Despite the huge size that these tumors can reach, large excision decrease local recurrence rate to almost zero. Studies have shown that well–differentiated liposarcoma located on the extremities does not require adjuvant therapy and overall they have prolonged survival and favorable prognosis.


Teratoma consists of tissues derived from all the three germ layers, and there may be presence of appendages as a representation of these germ layers as well. Teratomas of the ovary are known to occur in a fairly large number of women. These may be present clinically at a much later stage, permitting a limited treatment plan. Newer diagnostic techniques are always welcome in identifying these lesions. This case report discusses the case of a 40-year-old woman with a large teratoma in the right ovary and its diagnostic and surgical modalities. Teratoma is a true neoplasm arising from totipotential cells and made up of a variety of parenchymal cell types representative of more than one germ layer; usually all three . It is dMost of the tumours take up the features similar to dermoid cyst with ectodermal derivatives, and a majority of the malignant teratomas are squamous cell carcinomas with the other being carcinoids, adenocarcinoma, melanomas, and types of sarcomas . The cysAdvancement in imaging techniques has aided in the diagnosis, as well as adequate planning prior to the surgery. CT and MR imaging of pelvic regions are usually diagnostic of ovarian teratomas, wherein a CT shows typically fat-attenuated within a cyst, and an MR usually reflects the identification of the sebaceous components . Teeth m This case reported here represents a benign teratoma of the right ovary in a 40-year-old female patient reporting to the outpatient department. The provisional diagnosis was given as a right complicated cyst with an indication for dermoid cyst. Family history was noncontributory. The patient gave a history of menopause two years prior to the checkup. Further investigations of an ultrasonography (USG) revealed a 51 mm × 47 mm well-defined lesion with calcification of 8 mm noted in the right ovary, suggestive of a complicated ovarian cyst or a probable dermoid cyst offered as USG diagnosis. Commonly used marker for ovarian cancers, CA-125, was assessed, and normal levels of which suggested the absence of malignancy. An access to the lesion was established by a transverse incision over the abdomen, and the cystic tumour was obtained in toto. A cut section showed the presence of derivatives of all three layers, namely, teeth, sebaceous material, connective tissue, and fat. Grossing revealed the presence of mucinous material and few molariform teeth . The patA teratoma is defined as a benign tumour derived from all three germ layers, showing evidence of tissue formation from ectoderm, mesoderm, and endoderm and not being native to the tissue . Ovarian The malignant transformation of an ovarian teratoma has an incidence of less than 1% . This pr Various diagnostic modalities have been included to avoid misdiagnosis at the time of surgery initiating a dilemma in the surgeons mind. Radiographs may provide clues but only if there are calcified masses seen. Ultrasound is an important diagnostic tool which may aid the clinician in diagnosis, and a simple classification suggested on the echo patterns by Caspi et al. has made it possible to diagnose ovarian teratomas in more than 97% of the cases . Althoug Our case presents a 40-year-old woman with a history of menopause 2 years prior to the admission. The lesion in the right ovary was evident on the ultrasound and also presented calcified masses, which were indicative of teeth. Following surgery, the gross specimen revealed a cystic lesion and the presence of molariform teeth making it an obvious diagnosis. The histopathological examination revealed the presence of odontogenic like epithelium and developing teeth, which indicated that the lesion may have formed more teeth or a mandible if it would have been left undisturbed. Hence, an accurate diagnosis of such lesions with appropriate methods holds great importance in providing a correct view to the surgeon and aiding in complete removal of the lesion.


Sarcomas of the ovary can either be histologically pure or can represent components of a more complex tumor. Ovarian angiosarcomas are rare, and probably arise from carcinosarcomas, teratomas or the rich ovarian vasculature. To date, only two small case series have been published, one with four cases and the other with seven.A 41-year-old Saudi woman presented to our gynecological clinic with abnormal vaginal bleeding. The initial clinical diagnosis was left ovarian cyst. The results of the remainder of her abdominopelvic examination were normal. Peri-operatively, the left ovarian mass resembled a hemorrhagic solid tumor. It was sent for frozen sectioning, which revealed it was an undifferentiated neoplasm. The final histopathological examination showed a vascular neoplasm showing vasoformative arborizing channels of variable sizes and shapes lined by atypical endothelial cells with intact capsule. Areas of necrosis were seen, along with fused anastomosing solid vascular area. She was diagnosed as having an angiosarcoma of intermediate grade, International Federation of Gynecology and Obstetrics stage IA.Patients with ovarian angiosarcomas most commonly present with abdominal pain, however some patients present with distant metastases, often in the lungs. Spread beyond the ovary is present at the time of diagnosis in most reported cases, with disease progression within less than a year after diagnosis. Cases of advanced stage disease behave aggressively and demonstrate poor response to surgery and chemotherapy, with an overall poor prognosis. They have a tendency for local recurrence and metastases, and prognosis is hence poor; the reported five-year survival rate is 10 percent to 35 percent, however, cases confined to the ovary have survived up to nine years. Soft tissue sarcomas account for less than 1 percent of all malignancies. Gynecological sarcomas are rare and have poor prognosis. Among gynecological sarcomas, uterine sarcomas are the most common. Sarcomas of the ovary comprise less than 1 percent of ovarian malignancies, mostly seen as components of carcinosarcomas [Angiosarcomas comprise only 2 percent of all soft tissue sarcomas [et al. [Ovarian angiosarcomas are rare and probably arise from carcinosarcomas, teratomas or the rich ovarian vasculature. The development of a somatic malignancy, more frequently in the form of a sarcoma, is a well-known phenomenon in a germ cell tumor; however, the pathogenesis remains unclear. The sarcomatous element may appear in the primary tumor or manifest in recurrence or in the metastases. Dedifferentiation phenomenon, malignant transformation of certain mesenchymal elements within teratomas, origination from primitive germ cells and transformation of the blastematous stroma in yolk sac tumor are some of the proposed theories that have been put forward in the literature ,3. To daet al. , two aroet al. ,10. Receet al. ,12.Ovarian angiosarcomas are usually unilateral and can occur at any age ; with rare exceptions it is a disease found in premenopausal women and most cases are reported in women of childbearing age (<40 years). Only three cases have been reported in patients of postmenopausal age . The mosMore than half of the cases were disseminated at the time of diagnosis and only a few of them were detected at stage I, with disease progression of less than a year. Cases of advanced stage disease behave aggressively and demonstrate poor response to surgery and chemotherapy, with an overall poor prognosis. Few cases have shown good response to a regimen of doxorubicin and ifosfamide.A 41-year-old Saudi woman, para 7+0, presented to our gynecological clinic with abdominal pain and abnormal vaginal bleeding for the last six months. Her menstrual history was unremarkable. She had a history of a cesarean section three years previously and appendectomy performed a year ago. On examination, a palpable pelvi-abdominal mass was detected. Her hemoglobin was 10.1g and random blood glucose was 80mg. liver and renal function test results were within normal limits. An ultrasound study of her abdomen showed a normal-sized uterus displaced to the right side by a left ovarian mass, 8cm at its maximum dimension. No free fluid was seen. The initial clinical diagnosis was one of left ovarian cyst. The rest of her abdominopelvic sonographic examination was normal. Peri-operatively the mass resembled a hemorrhagic solid tumor. The left ovarian mass was sent for frozen sectioning, which revealed it was an undifferentiated neoplasm based on one frozen section; whether it was of benign or malignant nature could not be ascertained. The final specimen sent for histopathological examination consisted of a left salpingo-oophorectomy and incisional biopsy of the right ovary. Grossly, the left ovarian mass measured 7.0×6.0×2.0cm and was brownish and hemorrhagic, with solid and cystic areas. Permanent sections of the left ovarian mass showed a vascular neoplasm with adjacent compressed ovarian parenchyma at the periphery Figures  and 4. TSarcomas of the ovary either can be histologically pure or can represent components of a more complex tumor. Mature cystic teratoma (MCT) is the most common type of ovarian neoplasm, accounting for 26.5 percent to 44 percent of all primary ovarian tumors . MalignaThe ovary is a common site for metastases and 5 percent to 10 percent of the ovarian malignancies are metastatic . Detailset al. [The presence of sarcoma cells in effusions is an exceedingly rare finding. Davidson and Abeler  reportedet al.  also repThe microscopic appearance of angiosarcoma can vary, from focal or diffuse vasoformative components with dilated, slit-like, papillary or interconnecting vascular spaces to solid components that may be spindle sarcomatous or epithelioid. Microcystic/tubulocystic areas, and fascicular or reticular patterns can be seen. A diagnosis may be difficult if the vascular nature of the neoplasm is not evident and there are more poorly differentiated areas exhibiting solid growth patterns. The solid component can mimic fibrosarcoma, leiomyosarcoma or malignant fibrous histiocytoma. Immunohistochemically they are positive for endothelial markers including CD31 and CD34  and factor VIII-related antigen. Focal positivity for smooth muscle actin, HHF35, pankeratin and epithelial membrane antigen (EMA) can be seen, however desmin is negative. The superiority of CD31 in terms of sensitivity and specificity for endothelial cells is well recognized, with reported sensitivity rates of 80 percent versus 62 percent in vasoformative and poorly vasoformative angiosarcomas, respectively. Angiosarcoma should be included in a panel in which an endothelial neoplasm is in the differential diagnosis. Factor VIII-related antigen is a marker of endothelial cell differentiation and its sensitivity decreases from 84 percent to 29 percent as the vasoformative areas become more solid and poorly differentiated .The differential diagnosis includes hemangioma, epithelioid hemangiomas, infantile hemangioendothelioma and metastatic angiosarcoma, embryonal carcinoma, melanoma, poorly differentiated carcinoma and leiomyosarcoma. Pure primary sarcomas other than angiosarcoma that have been described to occur in the ovary include fibrosarcoma, rhabdomyosarcoma, leiomyosarcoma, chondrosarcoma, osteosarcoma, and malignant peripheral nerve sheath tumor. Benign vascular neoplasms of the ovary are uncommon. Case reports of isolated hemangiomas  of the oThe behavior of somatic malignancies in MCT is not determined by their derivation from germ cells but by their phenotype, and is similar to that of their sporadic counterparts; therefore, treatment should target the somatic malignancy rather than the germ cell tumor.et al. [Such malignancies respond poorly to adjuvant chemotherapy. The most common chemotherapeutic regimen used to treat is a combination of doxorubicin and/or ifosfamide. The combination of mesna, doxorubicin, ifosfamide and dacarbazine (MAID) chemotherapy has also been reported, however the efficacy is still uncertain. More than 60 percent of all reported cases of ovarian angiosarcoma were detected at stages III and IV. They have a tendency for local recurrence and metastases, and prognosis is poor; the reported five-year survival rate is 10 percent to 35 percent . The suret al. ,4,5,25.Examination of multiple sections of an ovarian neoplasm, use of immunohistochemical studies in cases of poorly differentiated tumors and awareness of occurrence of angiosarcoma in ovary may result in more cases being identified and reported.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.The authors declare that they have no competing interests.All authors have contributed in the writing of the manuscript and all authors read and approved the final manuscript.


To report a case of ocular surface squamous neoplasia (OSSN) masquerading as superior limbic keratoconjunctivitis (SLK). A 62-year-old woman was referred with foreign body sensation, irritation, photophobia and decreased vision in the left eye. She was initially treated for 10 months with intermittent topical corticosteroids for a presumed diagnosis of SLK. She underwent excisional biopsy of the superior conjunctiva and was found, on histopathologic evaluation, to have OSSN with moderate to marked dysplasia. This is the first reported case of OSSN masquerading with signs and symptoms of SLK. Any ocular surface lesion refractory to standard medical treatment should raise suspicion for a malignant process and warrant further cytologic or histopathologic evaluation. On the other hand, superior limbic keratoconjunctivitis (SLK) presents as chronic superior bulbar conjunctival injection and thickening, believed to be associated with mechanical soft tissue microtrauma. In either case, patients can present with similar symptoms, including foreign body sensation, irritation and redness.Conjunctival ocular surface squamous neoplasia (OSSN) lesions are described as papilliform, leukoplakic or gelatinous, appearing mostly at the limbus. These lesions are considered OSSN has been reported to present as corneal ulcer,A 62-year-old woman was referred with chronic redness, irritation and photophobia of the left eye for several months. She had no prior history of eyelid surgery or trauma. Her past medical history was significant for hypothyroidism. Multiple treatment regimens, including artificial tears, topical corticosteroids and bandage contact lens, had been tried. On examination, the best-corrected visual acuity was 20/20 in both eyes. Her left superior bulbar conjunctiva was injected with engorged and tortuous episcleral vessels . In addiPathologic examination with standard hematoxylin and eosin stains revealed diffuse thickening of the epithelium, with focal areas of dyskeratosis, atypia, pleomorphism, prominent nucleoli and clumped chromatin . These cSLK is characterized by a fine papillary reaction on the superior tarsal conjunctiva, injection and thickening of the superior bulbar conjunctiva, and superior corneal filamentary keratopathy.The location of OSSN is typically in the interpalpebral fissure,The transformation of the stratified epithelium into squamous carcinoma as a result of mechanical irritation has been described in the conjunctiva as a result of a poorly fitting prosthesis,OSSN presenting as cornea ulcer,1 chronic blepharoconjunctivitis,2 pterygium,3 necrotizing scleritis4 and sclerokeratitis5 have been reported in the literature and this is the first report of OSSN developing from or masquerading as SLK. In our case, the initial diagnosis of SLK was made based on the location of the lesion, classic slit-lamp appearance, clinical symptoms and patient’s associated past medical history of thyroid dysfunction.


Schwannoma is a neurogenic benign tumour arising from the proliferation of Schwann cells present in the peripheral nerve sheath of myelinated nerves. This proliferation can hypothetically appear in every anatomic region of the human body, but the nerve sheath tumors rarely occur within the larynx. In this paper the authors discuss the case of a 74-year-old female who presented to Emergency Unit (EU) for an important acute respiratory distress. Airway flexible endoscopy revealed a bulky mass of the aryepiglottic fold measuring 3.5 cm in diameter. The patient underwent tracheotomy and a single-step surgical excision treatment of the mass which was recognized as a schwannoma at pathological examination. Tracheotomy was closed 2 weeks postoperatively. After 18 months of followup, the patient is alive and free of disease and her voice had improved markedly. Schwannomas were first described in 1908 by Verocay, who termed them neurinomas. In 1940, Stout recognized the schwannian derivations of this neurogenic tumors and called the same entity schwannoma. Benign neurogenic tumors include neurofibromas and schwannomas . SchwannSymptoms of this lesion are related to mass effect; they include hoarseness, globus sensation, sore throat, odynophagia, dysphagia, dyspnoea, stridor, and dysphonia. Symptoms progress over months to years. Stridor and dyspnoea are the late findings . At leasSurgery is the therapy of election for this benign radioresistant tumor: they are well-circumscribed and encapsulated submucosal tumors that are extrinsic to nerve fascicles and thus are easily removed. Recurrence can follow an incomplete resection. Malignant transformation is extremely rare  and is oWe describe a case of schwannoma of the aryepiglottic fold presenting with rare clinical presentation as acute respiratory distress; diagnostic findings and therapeutic measures adopted are discussed.pCO2 82.9 mmHg,  pO2 52 mmHg; chest X-ray was negative for pulmonary disease. Family history was negative for remarkable diseases. Patient's past medical history included surgical excision of a cutaneous melanoma followed by chemotherapy (presently NED), carotid thromboendoarterectomy, a surgically treated CNS meningioma with implantation of ventriculo peritoneal shunt. Traumatic lesion of vocal cords, causing mild persistent dysphonia, was reported to have occurred during previous intubation for brain surgery. In the EU, the patient underwent a flexible endoscopic evaluation of the larynx that revealed a bulky mass on the left aryepiglottic fold with the involvement of the glottic space and consequent almost complete obstruction of the laryngeal respiratory space. No palpable lymph nodes were present.A 74-year-old female was referred to Emergency Unit (EU) of our hospital for an acute dyspnoea with 1-month history of progressive globus sensation; the patient was admitted to the Pneumology Department for evaluation of an important hypercapnic respiratory failure. Arterial blood gas analysis showed pH 7.35,  The patient was transferred to the Otolaryngology Department because of acute dyspnoea, and emergency tracheostomy was performed followed by incisional biopsy of the lesion. Intraoperatively, the mass was found to be smooth, pedunculated, and 3.5 cm in diameter. The histopathological examination of the incisional biopsy sample reported nonspecific fibrous submucosal tissue.CT scan demonstrated a round, heterogeneously enhancing mass, measuring 3.5 × 3 × 2.8 cm, which extended from the left aryepiglottic fold to the false vocal cord .A second single-step surgical excision was then decided. The left false vocal cord was incised with a sickle knife, and a microflap was dissected; blunt dissection revealed an encapsulated, soft tissue mass. The tumor was completely excised. Macroscopically the surgical specimen consisted of a well encapsulated tan-coloured tumor mass, measuring 4 × 2 cm. At microscopic examination, the tumor showed densely cellular areas with sheets of spindle cell palisading around amorphous matrix (Antoni A pattern) and less cellular areas with spindle cells in myxoid stroma and disorganized distribution (Antoni B pattern) . Tumor cTwo weeks postoperatively, the patient reported that her globus sensation, chronic throat clearing, and shortness of breath had disappeared. The tracheostomy was closed. One month after surgery, she was reevaluated in the outpatient clinic by flexible fiberoptic laryngoscopy that revealed a complete healing of the vocal cord, minimal erythema, and no edema. At 18-month followup, the patient presented no endoscopic evidence of tumor recurrence and her voice had improved markedly .In 1908, Verocay was the first to describe the tumors deriving from Schwann cells [Since then, few cases have been reported in literature to date accounting for less than 0.1% of all benign tumors of the larynx  Table 1Table 1.Symptoms are related to the mass effect of a slowly growing tumour including hoarseness and foreign body sensation in the throat. As the tumor expands, it may cause dyspnoea and stridor ; as theyComputed tomography (CT) scans recognize small schwannomas as homogeneously enhancing masses. Tumors larger than 3 cm in size are seen as masses with slightly heterogeneous contrast enhancement, suggestive of central necrosis surrounded by a peripheral enhancing ring. CT scan is used to define the extent of the lesion and its anatomic relationships . The dif2 laser has been reported with no recurrence [The treatment of choice in neurogenic tumors is a complete surgical excision that can be achieved endoscopically or through external approach depending on the localization and size of the lesion . For bulcurrence . Howevercurrence . Malignacurrence .


Traumatic macular holes (TMH) are well-known complications of ocular contusion injury. Spontaneous closure occurs in approximately 50% of cases, but rarely after the age of thirty. We report a case of spontaneous closure of a full thickness macular hole due to a blunt trauma and we suggest possible mechanisms for this closure.A 50-year-old Greek woman was referred with a history of reduced best-corrected visual acuity after blunt trauma to her right eye. Diagnosis was based on fundoscopic, optical coherence tomography as well as fluorescein angiography findings with follow-up visits at two days, 20 days and five months. Fundoscopy revealed a full-thickness TMH with a minor sub-retinal hemorrhage and posterior vitreous detachment. The presence of a coagulum in the TMH base was observed. Subsequently, TMH closure was observed.The clot in the TMH base, potentially a hemorrhage by-product containing a significant quantity of platelets, may have simulated the clot observed after autologous serum use, thus facilitating a similar effect. This may have stimulated glial cell migration and proliferation, thus contributing to spontaneous hole closure. Commotio retinae, diffuse retinal edema, retinal hemorrhage, vitreous hemorrhage, choroidal rupture, photoreceptor injury, and macular holes (MH) are well known complications of ocular contusion injury. According to relevant literature, the frequency of traumatic macular holes (TMH) is between 1% and 9% .A 50-year-old Greek woman was referred to us with a history of reduced best-corrected visual acuity (BCVA) after blunt trauma to her right eye. Past medical and ocular history was unremarkable. Her BCVA one hour after the incident was hand movements at 10 cm. An anterior chamber examination revealed a round pupil with no signs of hyphema or iridodialysis. Fundoscopy revealed a full-thickness TMH with a minor sub-retinal hemorrhage and posterior vitreous detachment , despite a foveal hemorrhage. Fluorescein angiography (FA) revealed masking in the fovea, progressive staining peripheral to the masking area and a central window defect Figure . OCT exaSeventeen days later, her BCVA had improved to 7 Early Treatment Diabetic Retinoapthy Study (ETDRS) letters at 4 m. Fundoscopy and OCT scans revealed TMH closure and adjacent pigment dispersion Figures , 2. FourTMH is a rare complication of blunt trauma; contrary to idiopathic MHs, it usually has a lamellar configuration . Spontanet al. [Yamashita et al.  proposedOur patient shared common features with similar previously reported cases; an observed full-thickness TMH, and a limited BCVA improvement , despiteIn our case, the clot in the TMH base, potentially a hemorrhage by-product containing a significant quantity of platelets, may have simulated the clot observed after autologous serum use, thus facilitating a similar effect. This may have stimulated glial cell migration and proliferation, thus contributing to spontaneous hole closure.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.MBN was involved in data acquisition and manuscript drafting. CS was involved in manuscript drafting. IT was involved in data acquisition and manuscript drafting. SA revised the report critically for important intellectual content. TR was involved in data interpretation and revised the report critically for important intellectual content. SAD gave final approval of the version to be published. All authors read and approved the final manuscript.


Benign oropharyngeal tumors are far less common compared to malignant tumors. Oropharyngeal papilloma is present in adults. Squamous papillomas are exophytic masses of oral cavity. When they occur on the palate they are most often asymptomatic and benign. Pedunculated squamous papillomas usually arise from the soft palate, tonsil, or the epiglottis. These lesions can sometimes prove to be fatal. A case of pedunculated squamous papilloma, arising from the laryngopharynx, the clinical presentation, the site of origin, and the size of the lesion were quite unusual. The narrow base of the pedicle made the intraoral excision possible. But here, we present a case of a 10-year-old boy who had history of dysphagia of 3-year duration for solid food and he was comfortable only in squatting position the reason being squamous papilloma of uvula  atypically because of astonishing length of pedicle (2.3 inches).  Benign oropharyngeal tumors are far less common compared to malignant tumors. Oropharyngeal papilloma is present in adults . SquamouA 10-year old boy presented with 3 years of unusual dysphagia. He also reported choking sensation while eating solid food and was comfortable only in squatting position. He had a history of elongated pedunculated lesion. The patient had difficulty in swallowing food for 7 years because of local irritation in throat and dry cough. There was no relevant history of throat pain, fever, and any other infection. On throat examination extending from uvular tip a fine strand of tissue over 2.3 inches in length ending in a small disc-like mass-pedunculated (3 × 2 cms) basal part of uvula appeared to be normal. This caused difficulty in swallowing . Excision was simple see .Grossly. On examination the disc-like mass revealed rough and papillary projections giving a provisional diagnosis of papilloma  is a benign proliferation of the stratified squamous epithelium, which results in a papillary or verrucous exophytic mass. The lesions were softened/flaccid in 66.7% of cases and a pedunculated attachment was seen in 75% of the lesions .Male to female ratio is 1 : 1.5. The mean age was 33 years, with the majority in the 2nd, 3rd, and 4th decades. The sites commonly affected by benign neoplasms were the palate, tongue, upper lip, and buccal mucosa, in a descending order. 4% of squamous papilloma was common benign oral soft tissue masses. The most common location for squamous papilloma was the palate and tongue [Malignant oropharyngeal tumors are far more common compared to benign tumors. Oropharyngeal papilloma is typically present in adults. Benign oropharyngeal tumors are far less common compared to malignant tumors. Oropharyngeal papilloma is typically diagnosed most often in adults. The papilloma is usually solitary and enlarges to a maximum size of about 0.5 cm [In 464 oral squamous cell papillomas, 34.3% of cases were located on the palatal complex , but only 4.2% of cases were located on the uvula. Papilloma was greater than one cm in length, which occurs in less than 25% of cases of oral papillomas. Therefore, it is probable that the greater the length of the uvular lesion, the rarely [They are most often benign and asymptomatic. But in our case the patient presented with dysphagia-irritation of throat-atypical . SquamouPathogenesis is related to human papilloma virus, but there is controversy regarding its viral origin. In our case section shows koilocytes . AverageSquamous cell papilloma is a benign tumor with a rare entity arising on uvula which caused unusual dysphagia (symptomatic) of long duration, and patient was comfortable only in squatting position; it revealed fine strand of tissue measuring 2.3 inches terminating with a mass measuring 3 × 2 cms, papilloma, and on section it revealed presence of koilocytes. Hence with the above features it is a rare entity.


Ganglioneuromas (GNs) are well-differentiated, slow-growing, benign tumors that are quite rare and usually found in the posterior mediastinum and retroperitoneum. They are composed of ganglion and Schwann cells and their origin remains in dispute. GNs have been reported as intraosseous lesions, such as in temporal and orbital bones. There are rare reports of intracranial lesions, mostly in the pituitary fossa. Most GN patients are children and are clinically asymptomatic. Diagnosis of GN requires histopathologic evaluation since no specific clinical or radiologic diagnostic features have been identified. We report the case of a 35-year-old man with recurrent sinusitis whose radiologic workup revealed a lytic right sphenoid wing lesion with microcalcifications. He underwent gross-total resection of the lesion and the pathologic findings were diagnostic of ganglioneuroma. To the best of our knowledge, this is the first reported case of sphenoid wing GN. The nature and origin of this tumor are discussed, and the GN literature is reviewed.  Ganglioneuromas (GNs) are benign tumors of the peripheral nervous system that are seldom seen in the cranial bones or intracranially. They are composed of mature ganglion cells, occasional satellite or “capsular” cells, unmyelinated axons and Schwann cells . While tWhether arising in the soft tissue or bone, GNs can increase in size and cause compression of adjacent tissue . IntraosAdditionally, familial disposition as well as an association with neurofibromatosis, Turner Syndrome and multiple endocrine neoplasia syndrome Type-B has been reported . Rarely These slow growing benign tumors are not always symptomatic but if symptomatic, it is usually due to compression of autonomic or somatic peripheral nerves. In this paper, we present a right sphenoid wing GN that was found incidentally in a 35-year-old man. A 35-year-old man who had recently undergone septoplasty and bilateral turbinate reduction for frequent episodes of sinusitis was found to have a lytic lesion with smooth borders in the right sphenoid wing on preoperative computerized tomography (CT) scan. After surgery, he was referred for neurosurgical evaluation of the asymptomatic mass. No abnormality was found on the general or neurological examination. Cranial nerve examination in particular showed that facial sensation was intact. CT scan of the head revealed a fibro-osseous, well-demarcated lesion in the right sphenoid wing with remodeling and sclerosis of the adjacent bone A. MagnetDue to uncertainty regarding the nature of this lesion, the patient elected for excisional biopsy. He underwent a right cranio-orbital approach for microsurgical gross total resection of the lesion. Extradural dissection was performed and the dura was reflected from the greater wing of the sphenoid bone to expose the lesion. The V2 and V3 branches of the trigeminal nerve were identified, and the lesion was found near the foramen rotundum. The trigeminal ganglion and/or trigeminal recess (Meckel’s cave) was not exposed since the lesion was not reaching posteriorly. The V2 branch of the trigeminal nerve was severely compressed in the foramen by the tumor. However, no nerve infiltration was observed. Nerve decompression was achieved by microsurgical resection of the tumor. Histologically, the excised tissue revealed a well-differentiated intraosseous neoplasm, replacing the bone marrow and consisting of a large number of mature ganglion cells in a moderately cellular stroma of spindle cells a – f. ThGanglioneuroma (GN) is classified as a benign well-differentiated neoplasm of the peripheral nervous system (PNS) . GNs areGNs are usually seen before the second decade of life and rarely after the sixth . The medConsidering the radiological findings, age bracket, and the location of the lesion, our preoperative diagnosis favored intraosseous meningioma. However, microscopic tissue examination reveals typical features of a ganglioneuroma. GNs lack cellular atypia, mitotic activity, and necrosis , and oveIn summary, cranial ganglioneuromas are extremely rare. Being benign tumors, gross-total surgical excision is considered to be curative. However, clinical follow up of the patient is recommended since rare cases of malignant transformation have been reported. In this case report, we present the first case of ganglioneuroma involving the sphenoid wing. The authors thank Traci Niesen HT (ASCP), Sally A. Drew MT (ASCP), and Linda A. Sebree HT (ASCP) for their excellent technical assistance, as well as Korise Rasmusson PhD and Gregory Kujoth, PhD, for their assistance with preparation of this report.


The Spigelian hernia or lateral ventral hernia is a rare clinical entity, characterized by a defect in the Seminular line. The etiology is mainly based on the anatomical features of the Spigelian aponeurosis and all those conditions that lead to increased intra-abdominal pressure , typical in the elderly.In our study, we report two cases of Spigelian hernia which appear in two patients, a 75-year-old female and a 71-year-old male, both had associated comorbidities. The female patient, suffering from hypertension and a history of diverticular disease which presents a strangulated hernia of the left abdominal wall, so, in the presence of intestinal sub-occlusion sintomatology, required emergency treatment. The second patient, suffering from hypertension, kidney stones and benign prostatic hyperplasia, reported abdominal discomfort with intense swelling, predominantly on the left of the abdominal examination. The second patient did not present complication and elective treatment was performed. The surgical technique adopted in both cases was the placement of hernioplasty with polypropylene mesh, a technique that allows a short hospital stay and early rehabilitation for the patient.


Background. Adult rhabdomyoma is a rare benign tumour with the differentiation of striated muscle tissue, which mainly occurs in the head and neck region. Twenty-six cases of multifocal adult rhabdomyoma are documented in the literature. Method. We report a 55-year-old male with simultaneous diagnosis of 7 adult rhabdomyomas and review the literature of multifocal adult rhabdomyoma. Result. Review of the literature revealed 26 cases of multifocal adult rhabdomyoma, of which only 7 presented with more than 2 lesions. Mean age at diagnosis was 65 years with a male to female ratio of 5.5 : 1. Common localizations were the parapharyngeal space (36%), larynx (15%), submandibular (14%), paratracheal region (12%), tongue (11%), and floor of mouth (9%). Besides the known radiological features of adult rhabdomyoma, our case showed FDG-uptake in (18) F-FDG PET/CT. Conclusion. This is the first case of multifocal adult rhabdomyoma published, with as many as 7 simultaneous adult rhabdomyomas of the head and neck. Rhabdomyoma, named by Zenker  in 1864,In this study we present a case of multifocal ARM with simultaneous diagnosis of 7 lesions. To our knowledge this is the first patient reported to have ARM in more than 3 locations. Moreover, we review the literature on multifocal ARM.A 55-year-old male presented with a 3-month-history of hoarseness and slight dysphagia. His past medical history was significant for tonsillectomy and spontaneous pneumothorax. Physical examination showed an impressive asymmetry of the soft palate due to a right parapharyngeal mass. Moreover, there was a soft mobile submandibular mass palpable on the right. Magnetic resonance imaging (MRI) was performed which showed a large, homogenous, well-circumscribed parapharyngeal tumor on the right with a maximal diameter of 8.5 cm, extending inferiorly to the submandibular gland. A second tumor of smaller size was seen in the opposite parapharyngeal space. Both tumors had a similar aspect slightly hyperintense to muscle on native T1 and T2 with a light homogenous contrast enhancement . A biopsSurgical removal of the parapharyngeal tumor on the right and the paraglottic mass on the left was performed, since they were responsible for the patient's symptoms. Both could be removed more easily than expected. The parapharyngeal mass was removed by a submandibular incision, from which the tumor could be released. Similarly, the paraglottic lesion could be removed endoscopically by an incision of the false vocal cords. Both tumors had a likewise appearance, brownish, soft, lobulated, with a smooth shiny surface . HistoloBizon et al.  describeAdult rhabdomyomas are rare benign tumors with the differentiation of striated muscle tissue. They are considered to be true neoplasms unlike cardiac rhabdomyomas that are regarded as hamartomas . Ninety The typical macroscopic description of the tumor is that of a soft, coarsely lobulated, tan-grey, well-circumscribed, or encapsulated lesion. The definitive diagnosis is mostly made histologically but several authors emphasize the utility of fine needle aspiration cytology in making the correct diagnosis. To confirm skeletal muscle differentiation immunohistochemical stains are necessary. Even though the histology of ARM is distinctive, it is often mistaken for a variety of other lesions, particularly granular cell tumor, as well as hibernoma, oncocytoma, and paraganglioma , 5.The treatment of choice is surgery since ARMs are well circumscribed and easily removable by blunt dissection over a small incision. However, a recurrence rate from 16% to 42% has been described , 12. No About 15% of the patients with ARM present with multifocal lesions .  Table ARM is a very rare tumor with predilection for specific areas of the male head and neck. It has typical radiological as well as macroscopic characteristics and it shows elevated FDG-uptake in (18) F-FDG PET/CT. Definitive diagnosis is made by fine needle aspiration cytology or final histology and requires immunohistochemical stains. It is important that correct identification of the ARM is made in order to avoid an unnecessarily aggressive resection, yet providing potentially curative therapy. Even though not much literature exists we only recommend surgery of lesions that are symptomatic or cosmetically disturbing. Removal is often possible by a small incision, since ARM is well circumscribed and not adherent to adjacent structures. The occurrence of multiple lesions  has to be considered, since most likely postoperative persistence of tumor lobules is the reason for the high recurrence rates documented in the literature. To our knowledge this is the only case published with as many as 7 simultaneous ARMs.


The purpose of this paper is to present sonographic and CT imaging findings of xanthogranulomatous cholecystitis (XGC) presented as Bouveret's syndrome, a very rare cause of gastric obstruction. While the patient's physical examination, upper GI endoscopy, and radiological findings all pointed to Bouveret's syndrome, CT differential diagnosis suggested either XGC or gallbladder carcinoma, and the final diagnosis was done histopathologically. Our paper aims to increase awareness in radiologically diagnosing XGC cases by introducing the possibility of existence of Bouveret's syndrome. Xanthogranulomatous cholecystitis (XGC) is an unusual disease, with an estimated incidence of between 0.7% to 13.2% of all cholecystitis cases, while the frequency is about 1.5% of all the cholecystectomies studied .Bouveret's syndrome, on the other hand, is also known as “gastric outlet obstruction” and is a rare condition representing fewer than 5% of the cases with gallstone ileus, which itself is a complication in only 0.3–4% of cases having cholecystolithiasis [Despite a few studies with small series in the literature, there is a lack of data about the exact prevalence of the Bouveret's syndrome accompanying XGC . In thisA 75-year-old man was admitted to emergency department with a 20-day history of worsening nausea, vomiting, hiccupping, obstipation, and inability to pass flatus. He had cachexia associated with a remarkable decrease in turgor and tonus. Physical examination demonstrated tenderness in the right upper quadrant. US was suboptimal due to the massive intestinal gas and uncooperative nature of the patient. A collapsed GB and a mass with a smooth curvilinear surface and posterior acoustic shadowing in the distal part of the duodenum were the only distinguishable findings . After aIntraoperatively, decompressed and edematous GB was seen to be fistulized to the second part of the duodenum. Duodenal and proximal jejunal loops were dilated. The gallstone causing obstruction was found in the last segment of the dilated proximal jejunal loops. Cholecystectomy and primary repair of the duodenum was performed and followed by the placement of an 8 French T-tube in the common bile duct. The gallstone was extracted from the jejunum with a simple enterotomy made 25 cm distal to the ligamentum Treitz followed by an antecolic gastroenterostomy. Histopathological examination of the GB came out to be xanthogranulomatous cholecystitis.XGC is an unusual form of chronic cholecystitis and predominantly seen in women between the ages of 60 and 70 years. Patients normally present with signs and symptoms of cholecystitis: right upper quadrant pain, vomiting, leukocytosis, and a positive Murphy sign. Slightly less than one-half of patients have a tender, palpable, right upper quadrant mass at physical examination. Some patients present with anorexia , 7. Our On US, XGC may appear as a moderate-to-marked thickening of the GB wall and hypoechoic bands or nodules within the thickened gallbladder wall. Other sonographic findings include disruption of the mucosal line, pericholecystic fluid, stones, and intrahepatic biliary dilatation. However, due to the decompression of the GB, US might have difficulty in showing the changes in the wall as in our patient. Clean or dirty acoustic shadowing from stones or air within the GB lumen, air in the bile ducts, dilatation of small bowel, and fistula can be detected sonographically . The onlCT may demonstrate low-attenuation foci in the gallbladder wall that corresponds to the hypoechoic nodules seen at US with an irregular, sometimes lobulated, and greatly thickened wall in XGC. The GB mucosa typically enhances with contrast material. CT more effectively demonstrates adjacent organ involvement and infiltration of the adjacent soft tissue and fat planes when compared with US. Occasionally, biliary dilatation is present and may be secondary to the presence of intraductal stones, hepatoduodenal ligament adenopathy, or in rare cases a coexistent malignancy of the gallbladder or bile duct. The preoperative diagnosis of XGC is also difficult with CT and most of the patients were diagnosed by an intraoperative frozen section examination or postoperative specimen , 6, 7. ABouveret's syndrome is frequently preceded by one or several episodes of acute cholecystitis resulting inflammation and adhesions facilitating the erosion of the gallstone through the gallbladder wall forming a cholecystoenteric fistula. The fistula can be cholecystoduodenal 60%), cholecystocolic (17%), cholecystogastric (5%), choledochoduodenal (5%) . Complic0%, choleCT can identify stones in the intestinal lumen, intestinal obstruction, pneumobilia, and sometimes the bilioenteric fistula in the Bouveret's syndrome. In our patient, who had no prior ERCP or surgery, the abdominal CT demonstrated pneumobilia, a big gallstone within the last two portions of the duodenum, and gastric dilatation. Bilioenteric fistula can be identified with different modalities such as an upper gastrointestinal barium study, CT, ERCP, MRCP , 10. In In conclusion, although the formation of firm adhesions and fistulas are not unusual in the natural course of XGC because of destructive inflammation, to the best of our knowledge, the presentation of the XGC as gallstone ileus as in our patient has not been demonstrated in the current literature. We believe the combination of these two entities can be an emergency in both clinical and radiological aspects and is a topic that all radiologists should be aware of.


Chronic expanding hematoma is a rare condition that develops after surgery, trauma, or injury. It can also develop at any location in the body in the absence of trauma. Clinical findings and various diagnostic imaging modalities can aid in the differential diagnosis of this condition. In general, hematomas are naturally reabsorbed and rarely cause serious problems. However, hematomas that develop slowly without a history of trauma, surgery, or bleeding disorders could be difficult to differentiate from soft tissue neoplasms. In the present case, we describe a patient, without any history or physical evidence of trauma, who exhibited a large chronic expanding hematoma in the retroperitoneal space that resulted in hydronephrosis because of the pressure exerted on the left ureter.A 69-year-old man presented to our hospital with a swollen lesion in the left flank. A mass, 19 cm in diameter, was detected in the retroperitoneal space by computed tomography. We suspected the presence of a chronic expanding hematoma, soft tissue tumor, or left renal artery aneurysm. Surgical treatment was performed. However, postoperative histopathological examination indicated that the mass was a nonmalignant chronic expanding hematoma. No recurrence was observed during a 2-year follow-up period.In patients without a history of trauma who present slowly growing masses, the differential diagnosis should include chronic expanding hematoma in addition to cysts and soft tissue tumors. Moreover, the use of magnetic resonance imaging and computed tomography is essential to differentiate between chronic expanding hematoma and soft tissue tumors. Hematomas can develop in many locations of the body as a result of trauma, surgery, or bleeding disorders. The diagnosis of this condition is based on medical history, physical findings, and the results of examinations involving various imaging modalities. Some hematomas persist as slowly expanding, space-occupying masses for months or years, and are termed as chronic expanding hematomas (CEH) . In geneA 69-year-old man with no prior history of medication or anticoagulant therapy first noticed a painless swelling in his left flank in 2005. This swelling gradually increased over 6 years, resulting in the formation of a mass in the left flank region. In June 2011, the patient presented to a local hospital after mild pain developed. The patient did not have any definite history of trauma or surgery that could have caused the left abdominal/back lesion. Computed tomography (CT) indicated the presence of a left retroperitoneal mass with left hydronephrosis because of external compression by the mass and a right atrophic kidney. A double J-stent was placed in the left ureter. The serum creatinine level changed from 0.72 mg/dL before double J-stent insertion to 0.53 mg/dL after the treatment.On physical examination at the Gunma University Hospital, a firm area of swelling with an approximate size of 20 cm × 15 cm was detected. Blood tests results indicated that the patient had mild anemia and a hemoglobin level of 10.7 g/dL. The patient’s coagulation profile and platelet count were normal. However, CT indicated the presence of a huge mass (20 cm × 15 cm × 13 cm) compressing the left kidney in the upward direction in the retroperitoneal space. Contrast-enhanced CT revealed that the mass was not uniform but included scattered calcification and a partly enhanced rim , sarcomas, actinomycosis, and inflammatory pseudotumors . Weiss eVarious imaging modalities have been used for the diagnosis of CEH. It has been stated that a dynamic CT scanning can detect a rim enhancement in the arterial phase in such cases, because granulation tissue with vascular channels is distributed within the hematoma capsule ,9. In thThe optimal treatment option for CEH is complete excision of the hematoma together with its fibrous capsule. Aspiration of the liquid or drainage could result in serious bleeding or recurrence ,13. HoweIn the present case, the left kidney was hydronephrotic. Therefore, a double J-stent was placed in the left ureter to aid in identifying and preventing injury to the left ureter. The double J-stent was removed 2 months postoperatively, and the hydronephrosis in the left kidney had resolved. To our knowledge, a hematoma of a comparable size as the one reported in the present case, accompanied with left hydronephrosis, has never been reported in the literature. Moreover, in the present case, the etiology of CEH was unclear and could not be determined during surgery. Although no recurrence was evident at the 2-year follow-up, it is essential to further follow-up the patient carefully.We reported the case of a patient with a huge CEH located in the retroperitoneal space that resulted in hydronephrosis. MRI may be useful for differentiating between CEH and malignant soft tissue tumors. Complete removal of the CEH resulted in resolution of hydronephrosis and no recurrence of CEH at the 2-year follow-up.Written informed consent was obtained from the patient for publication of this Case report and any accompanying images.CEH: Chronic expanding hematomas; CT: Computed tomography; MRI: Magnetic resonance imaging.The authors declare that they have no competing interests.TK was responsible for acquisition of data, drafting of manuscript and preparation of the figures and table. MH and MN operated the patient and revised manuscript. TS and KS was responsible for critical revision of the manuscript. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2490/13/60/prepub


A 58-year-old man presented with a six-month history of intermittent blood-stained posterior nasal discharge. Five years ago, he had a three-week episode of fitful light headaches. Nasal ventilation, olfactory sensation, and facial sensation were normal; there were no ophthalmological complaints. Coronal computed tomography (CT) scans revealed soft masses in the bilateral sphenoid sinuses with bone absorption. The patient underwent bilateral functional endoscopic sinus surgery and resection of right nasal papillary masses. Papillary masses and mucosa in both sphenoid sinuses were also removed. The mass in the left sphenoid sinus was diagnosed as two separate entities, one being a primary monophasic epithelial synovial sarcoma and the other an inverted papilloma, while the mass in the right sphenoid sinus was an inverted papilloma. After surgery, the patient underwent radiotherapy and chemotherapy. At the 50-month follow-up visit, there were no signs of recurrence. Synovial sarcoma (SS) accounts for approximately 8% of all soft tissue sarcomas and is the fourth most common type of sarcoma. SS can occur anywhere in the body, but it most often occurs in the paraarticular areas of the lower extremities. The head and neck region is the second most common site of involvement, accounting for up to 10% of cases . We repoA 58-year-old Chinese man visited our department complaining of intermittent blood-stained posterior nasal discharge for six months and a three-week episode of fitful light headaches, which occurred five years ago. The patient reported normal nasal ventilation, olfactory sensation, and facial sensation. He had no ophthalmological complaints. Physical examination showed a polypoid mass in the left middle nasal meatus and a papillary mass in the right posterior olfactory cleft. No enlargement of the cervical lymph nodes was discovered. Coronal computed tomography (CT) scans of the sinuses demonstrated soft masses in the posterior ethmoid sinuses and bilateral sphenoid sinuses with bone absorption . Tissue The patient underwent bilateral functional endoscopic sinus surgery and resection of the right nasal inverted papilloma. Intraoperatively, a polypoid mass was found in the left middle nasal meatus. There was mucus present in the posterior right and left ethmoid sinuses. No mass was discovered in the ethmoid sinuses; rather, only edema was present. Absence of tumor cells was confirmed by pathology. Papillary masses in bilateral sphenoid sinuses were also found. Radical excision of the papillary masses was performed. The mucosa of the sphenoid sinuses was also removed, while the sella turcica and lateral walls were kept intact. Pathologic examination with light microscopy revealed that the mass in the left sphenoid sinus actually consisted of two distinct parts. In one part, atypical proliferating epithelial cells arranged like irregular glands (adenocarcinoma-like) distributed among a fibrous substance were observed . In the The incidence of SS in the paranasal sinuses is very low, particularly in the sphenoid sinus. To our knowledge, this is the first reported case of primary MESS with inverted papilloma in the sphenoid sinus. Pathologically, SS is a distinct sarcoma composed of two morphologically different cell types—epithelial and spindle cells. There are four subtypes of SS based on the relative proportions of the two cellular components and the degree of differentiation as follows: MFSS; MESS; biphasic; poorly differentiated . ImmunohThe optimal treatment of SS is complete surgical resection with histologically clear margins; however, this cannot always be accomplished in head and neck cases because of the proximity of the tumor to vital structures . AdjuvanPatients with SS in the sphenoid sinus do not have a specific presentation in prophase and are often free of signs or symptoms. Our patient only had intermittent blood-stained posterior nasal discharge and fitful light headaches that may not even have been caused by the SS. Once patients develop severe headaches, visual impairment, or restricted eye movement, the SS has involved surrounding structures. Early nasal endoscopy and computed tomography scans are essential; however, neither the radiologist nor the other consultants can be certain if a destructive lesion is SS, a different neoplasm, or inflammation. Thus, early tissue diagnosis is essential. Additionally, other problems arise if these pathologies occur concomitantly. Further work must be done to refine the diagnosis, prognosis, and treatment of synovial sarcoma in the paranasal sinuses.


Unicentric CD is known to be usually of the hyaline vascular variant, plasma cell and mixed type of this form are quite uncommon. Malignancies are mainly associated with the multicentric form. We report a rare case of unicentric mixed variant CD evolving into intrabronchial, extramedullary plasmacytoma.Intrabronchial mass with consequential obstruction of the left main bronchus, left lung atelectasis and mediastinal lymphadenomegaly was detected by chest CT in our patient suffering from cough and hemoptysis. Pulmonectomy was performed, histopathological and immunhistochemical analysis of lymph nodes revealed mixed type of CD with interfollicular monotypic plasma cell proliferation. The intrabronchial mass consisted of monotypic plasma cells confirming plasmacytoma. Systemic involvement was not confirmed by further tests.Although malignancies more often present in multicentric CD that usually belongs to the plasma cell subtype, this case confirms the neoplastic potential of the rarest, unicentric mixed variant of CD. Castleman disease (CD) - also known as giant lymph node hyperplasia or angiofollicular lymph node hyperplasia - has a wide spectrum of appearances due to its heterogeneous pathological and clinical characteristics. It may appear in any part of the body as nodal or extranodal mass.Regarding the clinical presentation, unicentric  and multicentric (systemic) form can be identified. The unicentric type usually presents as a benign, asymptomatic disease affecting one single, or localized group of lymph nodes most often observed in the mediastinal region. Complete local surgical removal is curative in most cases.Multicentric CD frequently associated with HIV infection and Kaposi’s sarcoma, is more aggressive and usually high risk for malignant transformation into lymphoma or other malignant lymphoproliferative diseases. This foDuring the past decades, the group of CD subtypes has been expanded. Histopathological classification has just been modified and updated. AccordiThe hyaline vascular type was first reported by Benjamin Castleman in 1956[In contrast, the plasma cell variant of the disease is characterized by follicular hyperplasia and polyclonal interfollicular sheets of proliferating mature plasma cells. OccasioThere are casual cases described as “mixed type” in which the histopathologic features of both variants take place together suggesting a possible connection between the two forms.Two new histological categories have been described recently: the human herpesvirus 8 (HHV-8)–associated (plasmablastic) multicentric CD, which presents most frequently in immunosuppressed (particularly in HIV-positive) patients, and another subgroup, which is defined as not otherwise specified multicentric CD with non-specific pathological findings, that have similar histologic features to that of plasma cell variant but that cannot be classified into either HHV-8 associated or into the plasma cell variant type.Ninety percent of all cases belong to the hyaline vascular type of the disease, which is usually unicentric and has mostly an asymptomatic course but alternatively symptoms may occur due to localization. On the Helicobacter pylori positivity which was successfully eradicated. A few years ago she was diagnosed with asthma but did not take the medications prescribed.A nonsmoking 51-year-old obese woman presented with cough and hemoptysis to our center for further examinations. Her medical history included panic disorder, hysterectomy due to myoma and gastroesophageal reflux disease with concomitant 2+  erythrocyte sedimentation rate of 30 mm/h (reference range: 0–20 mm/h), a leukocytosis of 20000 cells/μL (reference range: 4000–10000 cells/μL) and slightly elevated level of uric acid of 474 μmol/L (reference range:0–340 μmol/L). Tumor markers also yielded normal results . Lung function test revealed dominantly restrictive ventilatory disorder : 2,5 L (85% predicted), forced expiratory volume in 1 second (FEV1):1,74 L (69% predicted), FEV1/FVC: 69%). Bronchoscopy could be performed only under general anesthesia because of glottic spasm. During the examination deformation of orifices with no endobronchial changes of the right side and - consistent with the CT findings- complete obstruction of the left main bronchus could be seen  revealed the same status as previously detected but this time with additional mediastinal lymphadenopathy.Patient underwent left side pulmonectomy with resection of the affected lymph nodes. Histological examination showed that the intrabronchial mass was comprised of monomorph mature plasma cells confirming endobronchial plasmacytoma. In peribronchial lymph nodes depleted germinal centres were revealed with expanded mantle zone showing the “onion skin” pattern due to the concentric layers of small lymphocytes. Interfollicular area consisted mainly of lymphocytes and interfollicular vascularisation. In other areas interfollicular sheets of mature plasma cells were shown with similar morphology as that of the plasma cells in the intrabronchial mass Figures  and5. ITree years after surgery the patient is asymptomatic, with no signs of recurrence of the disease.CD is usually reported as a rare benign disorder characterized by lymphocyte proliferation but several case reports published in the last decades proved its malignant potential. Malignancies are usually present with the multricentric form of CD including Kaposi sarcoma, B cell lymphoma, Hodgkin’s lymphoma and plasmacytoma. Other conditions were also observed accompanying mainly the multricentric form, e.g. POEMS syndrome , nephrotic syndrome, amyloidosis, connective tissue diseases and other autoimmune disorders,12. In tUnicentric cases of CD followed by lymphoma or solitary plasmacytoma are quite spare and were reported only in a few cases. ExtrameThe etiology of CD is not fully elucidated. The role of IL-6 cytokine may be one of the key components in the pathophysiology of the disease, its overexpression in germinal center cells of the lymph nodes was shown in the plasma cell type of CD. In miceThe definitive diagnosis of CD is based on histology. The results should be carefully evaluated as other lymphoproliferative disorders, especially lymphomas with plasmablastic features may resemble to CD regarding the histopathologic features. Plasmablastic lymphomas are also often associated with HIV or HHV8 infections similar to multicentric CD, and even EBV (Epstein-Barr virus) may occur in both entity,31. Low-The optimal treatment for patients with unicentric disease is surgical excision, which is usually curative irrespectively of the histological variant. If the disease is non-resecable, additional treatments - adjuvant steroid therapy and radiotherapy - were reported to be successful. TherapeIn cases of EMP, radiotherapy was accepted in general to be the first-line treatment based on the radiosensitivity of the disease. Small lesions may be cured with surgery without radiotherapy. Radiotherapy should be only considered when residual local disease can be detected. In our case surgical excision was part of the diagnosis. In respect of our patient, there is no evidence of benefit of adjuvant radiotherapy. Surgery was curative, the patient has no recurrence of the disease after 3 years of follow-up.Unicentric CD with concomitant neoplasm is a rare condition. Plasma cell or mixed type CD with monoclonal proliferation is even more exceptional. We reported a unique association of unicentric, mixed type CD with monoclonal plasma cell infiltration associated with extramedullary, intrabronchial plasmacytoma proving the neoplastic potential of this entity. According to previous findings plasma cell malignancy may have developed due to the elevated IL-6 levels, although we have not measured it in our patient. The significance of monoclonality present in plasma cell or mixed type of CD is still not clear; however according to our case, surgical removal of the lesions might be curative. Further studies are warranted to analyze the clinicopathological and cytogenetic characteristics of this condition and follow up more cases.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.CD: Castleman disease; HIV: Human immunodeficiency virus; IL: Interleukin; HHV: Human herpesvirus; CEA: Carcinoembryonic antigen; AFP: Alfa fetoprotein; CA: Cancer antigen; NSE: Neuron specific enolase; FEV1: Forced expiratory volume; FVC: Forced vital capacity; IL: Interleukin; VEGF: Vascular epithelial growth factor; EGFR: Epidermal growth factor receptor; FDC: Follicular dendritic cell.The authors declare that they have no competing interests.NE wrote the paper, VM and GL drafted the manuscript and revised it critically for important intellectual content, LT and GV, GB have been involved in drafting the manuscript, GV performed the hematological exams of the patient, AC performed the pulmonectomy, GB performed the chest CT exams and virtual bronchoscopy, JC and AS performed the histological examinations and immunophenotyping. All authors read and approved the final manuscript.