PURPOSE: To describe a rare case of conjunctival leiomyosarcoma initially diagnosed as a poorly differentiated squamous cell carcinoma. METHODS: Clinical, light microscopic, immunohistochemical, and ultrastructural findings are reported. RESULTS: A 56-year-old Caucasian woman was referred with a history of a progressive, rapidly growing mass in her left eye. Biopsy of the mass and histology yielded a first diagnosis of a poorly differentiated conjunctival squamous cell carcinoma. Orbital exenteration was performed 2 weeks later. Macroscopically, the exenteration specimen showed a soft mass completely involving the conjunctiva and extending to the eyelids and orbital structures. Histological examination revealed a malignant tumour composed of atypical, predominantly epithelioid large cells. Immunohistochemical and ultrastructural studies combined with the light microscopic findings contributed to clarify the diagnosis of epithelioid leiomyosarcoma. The patient was started on chemotherapy and radiotherapy, but died a few months later from widespread metastases. CONCLUSIONS: primary involvement of the orbit by a leiomyosarcoma is rare, but this eventuality should be considered in the differential diagnosis of rapidly growing orbital and conjunctival masses.

To investigate tumor necrosis factor-α (TNF-α), syndecan 1 and basic fibroblast growth factor (bFGF) balance in Crohn's disease (CD) strictures.

Objective: To verify the ultrafine conformation of term villi in diabetic and normal placentae. Villar dysmaturity and chorangiosis are considered the most frequent findings in diabetic placentae, but their histogenesis is still unclear. Study Design: We performed a morphometric study of 38 term villi in 5 diabetic placentae and of 37 term villi of 5 normal placentae in order to know the different extension of endothelial surface (VL), the maximum (D max) and minimum (D min) distance of the vessels from the basal membrane, as well as the exact thickness of basal membrane (MT BM). The villi were examined with transmission electron microscopy, and parameters were automatically acquired with the iTEM software (Soft Imaging System, Münster, Germany). Results: VL results were statistically higher in diabetic placentae than in normal ones. Also D max and D min were higher in diabetic disease. MT BM was not different in the two groups. Conclusion: Our findings show that, in the presence of chorangiosis, the vessel surface in diabetic placentae is higher than in normal group, but the vessels are randomly distributed in term villi. The basal membrane is not different in the two groups. Morphometric evaluation seems to be more accurate using ultrafine samples.

Hepatocellular carcinoma rarely metastasizes to the orbit. We report a 45-year-old male, HBV+, HIV+, with a past history of a liver transplant for ELSD (End stage liver disease) with hepatocellular carcinoma and recurrent HCC, who presented with painful proptosis and diplopia of the left eye. CT scans of the head revealed a large, irregular mass in the left orbit causing superior and lateral destruction of the orbital bone. Biopsy specimens of the orbital tumor showed features of metastatic foci of hepatocellular carcinoma. Only 16 other cases of HCC metastasis to the orbit have been described in literature and this is the first case in a previously transplanted HIV+, HBV+ patient. Key words : Hepatocellular carcinoma, orbit, HIV+, HBV+, liver transplant.

We report a case of benign multicystic mesothelioma in a 20-year-old woman referred because of amenorrhea. She underwent pelvic transabdominal ultrasound, which disclosed a micropolycystic appearance of the ovaries and a fluid collection in the pouch of Douglas. Tumor serum markers revealed an increase in CA19.9. Abdominal and pelvic computed tomography scans confirmed the presence of ascites. Laparoscopy disclosed small, thin-walled, translucent cysts in the Douglas cavity. The cysts were free-floating in a yellowish, sticky, gelatinous material. Microscopically, cystic lesions showed mesothelium-lined cystic spaces surrounded by a delicate thin fibrovascular wall. With immunohistochemistry, the tumor cells were strongly positive for cytokeratin and calretinin. These aspects were suggestive of benign multicystic mesothelioma. Electron microscopy confirmed the mesothelial nature of this tumor. Serial evaluation of the CA19.9 concentration showed a progressive decrease in the serum marker in the normal range. The patient is now well and symptom-free with no recurrence 24 months after surgery. The association between benign multicystic mesothelioma and increased CA19.9 serum concentration has been described only once, in a man. To our knowledge, this is the second case of benign multicystic mesothelioma associated with increased CA19.9 serum concentration and the first diagnosed in a woman. In the present case, a minimally invasive laparoscopic approach enabled not only histologic diagnosis of benign multicystic mesothelioma but also its surgical treatment. Although benign multicystic mesothelioma is a rare pathologic entity, it is important that sonologists include it in the differential diagnosis of diseases that manifest with ascites. Furthermore, all surgeons should be aware of the macroscopic and laparoscopic appearance of the lesion, and its generally benign course.

AIM: To evaluate mucosal baseline mRNA expression of tissue transglutaminase 2 (tTG2), interferon gamma (IFNγ), toll-like receptor 2 (TLR2) and Myeloid Differentiation factor 88 (MyD88) in patients with microscopic enteritis (ME). METHODS: We retrospectively enrolled 89 patients with ME of different etiology, which was defined within a 2-year mean period of follow-up. Baseline histological examination was performed on Hematoxylin-Eosin stained sections and CD3 lymphocyte immunohistochemistry was used for intraepithelial lymphocyte count (IELs). ME was defined according to the criteria of Bucharest Consensus Conference. For each patient, formalin embedded biopsy samples of the duodenum referred to the period of ME diagnosis were retrieved. Real-time polymerase chain reaction (RT-PCR) was used to detect the amount of mRNA coding for tTG2, IFNγ, TLR2 and MyD88, and the quantity was expressed as fold change compared to controls. Control group was represented by duodenal normal specimens from 15 healthy subjects undergoing endoscopy for functional symptoms. Comparisons among continuous variables were performed by One way analysis of variance (ANOVA) and Bonferroni's test. The χ(2) test was used for categorical variables. Pearson's test was used to evaluate correlations. Receiver operating curves were drawn for all four markers to estimate sensitivity and specificity in discriminating the development of CD and GS. RESULTS: After a period of follow up of 21.7 ± 11.7 mo, the following diagnoses were achieved: gluten related disorders in 48 subjects (31 CD; 17 GS) and non-gluten related ones in 41 (29 Irritable Bowel Syndrome - IBS; 12 Others). CD patients had the highest tTG2 levels (8.3 ± 4.5). The ANOVA plus Bonferroni analysis showed that CD > Other ME > GS = IBS > negative controls. A cut off value of 2.258 was able to discriminate between CD and GS with a sensitivity of 52.94% and a specificity of 87.1%. Additionally, CD patients had the highest IFNγ levels (8.5 ± 4.1). ANOVA plus Bonferroni demonstrated CD > Other ME > GS = IBS > negative controls. A cut off of 1.853 was able to differentiate CD and GS with a sensitivity of 47.06% and a specificity of 96.77%. Patients with non gluten-related causes of ME exhibited the highest TLR2 levels (6.1 ± 1.9) as follows: Other ME > CD = GS = IBS > negative controls. TLR2 was unable to discriminate CD from GS. Patients with CD overexpressed MyD88 levels similarly to non gluten-related causes of DL (7.8 ± 4.9 and 6.7 ± 2.9), thus CD = Other ME > GS = IBS > negative controls. A cut off of 3.722 was able to differentiate CD from GS with a sensitivity of 52.94% and a specificity of 74.19%. IELs count (15-25 and more than 25/100 enterocytes) strongly correlated with mRNA levels of all tested molecules (P < 0.0001). CONCLUSION: Our results confirm that a single marker is unable to predict a discrimination among ME underlying conditions as well as between CD and GS. Mucosal high levels of tTG and IFNγ mRNA may predict the development of CD more than GS with high specificity, despite an expected low sensitivity. TLR2 does not discriminate the development of CD from GS. MyD88 levels indicate that intestinal permeability is more increased when a severe intestinal damage underlies ME in both gluten related and unrelated conditions. Therefore, the results of the present paper do not seem to show a clear translational value. KEYWORDS: Celiac disease; Gluten sensitivity; Interferon gamma; Microscopic enteritis; MyD88; Tissue transglutaminase; Toll-like receptor 2

Melanomas of the oral cavity are extremely rare. Their rarity and their independence on exposure to UV radiation make them particularly interesting. The authors analyzed an oral multiphasic melanoma composed by a nodular nonpigmented ulcerated central region, a nodular ulcerated pigmented area, a pigmented nonulcerated region, and an area similar to a dysplastic nevus. They determined the expression of some genes involved in the differentiation and cellular transformation in morphologically different regions of melanoma. All these areas were also analyzed by electron microscopy. The various regions composing the melanoma expressed genes involved in melanogenesis and melanoma progression in a different manner. Electron microscopy observation of ultrathin sections of each region evidenced ultrastructural differences, being the cellular architecture more compromised in the most aggressive parts of the neoplasm. This pilot study identified morphological, molecular, and ultrastructural differences that characterize each region of the multiphasic melanoma.

A 65-year-old woman presented with a long standing, progressive exophthalmos of the right eye. Her medical history was significant for Churg Strauss syndrome, and was treated with immunosuppressive therapy. She had undergone two previous orbital biopsies showing inflammatory reactive lymphoid hyperplasia. A diagnosis of orbital inflammation in Churg-Strauss syndrome was suspected, and the immunosuppressive therapy was increased. Because of the lack of response to therapy, a further biopsy was performed, by lateral orbitotomy approach. Biopsy of the mass revealed a granular cell tumor composed of S-100 positive cells with an acidophilic granular cytoplasm and peripheral lymphocytic infiltration. A granular cell tumor, which is very rare in the orbit, should be considered in the differential diagnosis of orbital tumors, and if suspected, an excisional biopsy must be undertaken. Typical histopathological aspect of the granular cell tumor is characterized by the presence of S-100 positive closely packed polygonal cells with a granular cytoplasm

We report three cases of proptosis, in children aged 6, 10 and 12, whereby in all cases the first clinical, radiologic and ultrasonographic diagnosis was lymphangioma, while the final anatomopathological diagnosis was rhabdomyosarcoma. In presence of a rapidly worsening exophthalmos or eyelid swelling in a child, an early correct diagnosis is very important. Imaging techniques play a very important role in the diagnosis, but are often inconclusive and an excisional biopsy (if feasible) must always be considered.

We report three cases of proptosis, in children aged 6, 10 and 12. In all cases the first clinical, radiologic and ultrasonographic diagnosis was lymphangioma, while the final anatomopathological diagnosis was rhabdomyosarcoma. In presence of a rapidly worsening exophthalmos or eyelid swelling in a child, an early diagnosis is very important. Imaging techniques have a very important role in the diagnosis, but are often not conclusive and a excisional biopsy (if feasible) must always be considered.