Unusual locations of hydatid disease: A 10-year experience from a tertiary reference center in Western Turkey.

INTRODUCTION: Hydatid disease is an endemic parasitic infection caused by Echinococcus granulosus mostly seen in the Mediterranean countries. The most affected organ is the liver, however hydatidosis can be found anywhere in the human body. METHODS: The records of patients who were diagnosed with hydatid disease in our hospital from December 2005 to February 2016 were analyzed retrospectively. The cases were evaluated and recorded depending on their gender, age and the localization of the cysts. RESULTS: A total of 329 patients diagnosed over a 10-year period were included in our study. There were 202 females (61.4%) and 127 males (38.6%). The hydatid cysts were located in the liver in 257 (78.1%) patients and in unusual locations in 72 (21.9%) patients. The most common unusual site for hydatid cysts was the spleen followed by bones, central nervous system, soft tissue, the kidney and the gall bladder. Amongst these 72 patients who had hydatid cysts in unusual locations; 33 patients had concomitant liver hydatidosis, whereas 39 patients had primary involvement of unusual sites. Two patients with malignancies also had hydatid cysts in different locations. CONCLUSION: Hydatid disease affects many organs in the body and therefore it can pose a major diagnostic dilemma and it may mimic other entities. In endemic areas, a differential diagnosis of hydatid disease should be considered for cystic masses in any anatomical location.

[Evaluation of clinical and laboratory findings of adult visceral leishmaniasis cases]. / Eriskin viseral leysmanyaz olgularinin klinik ve laboratuvar bulgularinin degerlendirilmesi.

Visceral leishmaniasis (VL, kala-azar) is a zoonotic infection caused by Leishmania species which are transmitted to humans by the bites of infected female phlebotomine sandflies. Leishmania infantum is the responsible species of VL in Aegean, Mediterranean, and Central Anatolia regions of Turkey mainly observed sporadically in pediatric age groups. The aim of this study was to evaluate the clinical and laboratory findings of adult patients with VL who were admitted to our hospital. A total of 10 patients (3 female, 7 male; age range: 18-67 years, mean age: 39.3 ± 16.51) followed in the infectious diseases clinic of the hospital between 2000 and 2013 were evaluated retrospectively. The diagnosis of VL was based on the presence of appropriate clinical and physical examination, as well as biochemical findings, positive serological test results (indirect fluorescent antibody test, and rK39 rapid antigen test) and/or detection of amastigote forms of parasite in the bone marrow aspiration samples. Of the cases three were diagnosed with both bone marrow and serology positivity, five with bone marrow positivity and one of each only with liver biopsy and positive serology result. Time interval from onset of the symptoms until the establishment of the specific clinical diagnosis was ranged from 2 to 12 weeks. The most frequent initial symptoms were fever, fatigue and abdominal distension. None of the patients had immunosupressive conditions such as HIV infection, corticosteroid use, immunosupressive treatment, or transplantation. All the patients were from Aegean region and six were living in rural areas. In all cases, hepatosplenomegaly, increased erythrocyte sedimentation rate, albumin/globulin ratio inversion, anemia, leukopenia and among nine cases trombocytopenia were detected. In one case acute renal failure has been developed before treatment and the patient was admitted to dialysis program. Bacterial superinfection occurred in two cases. Patients were treated with either meglumine antimonate (Glucantime(®), 20 mg/kg/day, intramuscularly for 28 days) or liposomal amphotericin B (3 mg/kg/day, intravenously for the first 5 days, then on 14(th) and 21(st) days) according to the availability of drugs. During one year follow-up period all of the patients improved and no recurrence was seen in any patient. In conclusion, among adult patients with fever uncontrolled within 2-week course of ampiric antibiotic treatment without any infectious focus or malignancy, VL should also be considered.

Cysteinyl 1 receptor antagonist montelukast, does not prevent peritoneal membrane damage in experimental chronic peritoneal dialysis model in rats.

BACKGROUND/AIMS: Continuous ambulatory peritoneal dialysis (CAPD) induces structural changes in the peritoneal membrane such as fibrosis, vasculopathy and angioneogenesis with a reduction in ultrafiltration capacity. Leukotriene (LT) receptor antagonists have been found to be effective to prevent fibrosis in some nonperitoneal tissues. The aim of this study is to investigate the possible beneficial effect of montelukast, a LT receptor antagonist, on peritoneal membrane exposed to hypertonic peritoneal dialysis in uremic rats. METHODS: Of the 48 male, 5/6 nephrectomized Wistar rats 29 remained alive and were included in the study. These studied rats were divided into 3 groups: Group I (n=7) was the control group, Group II (n=8) was treated with 20 ml hypertonic PDF intraperitoneally daily and Group III was treated with montelukast and similar PDF treatment protocol. The morphological and functional changes in the peritoneal membrane as well as cytokine expression were compared between groups. RESULTS: Submesothelial thickness and the severity of the degree of hyaline vasculapathy were more prominent in group III when compared to group I. There were no significant differences between group II and other groups in terms of submesothelial thickness and the severity of the degree of hyaline vasculapathy. Increased expressions of TGF-ß and VEGF in parietal peritoneal membrane were found in group II and group III when compared to group I. The amount of TGF-ß and VEGF expression were similar in group II and group III. CONCLUSION: This study suggests that montelukast treatment does not prevent the peritoneal membrane from deleterious effects of hyperosmolar PDF in the uremic environment.

Effects of mucosal TNF-alpha levels on treatment response in Crohn's disease patients receiving anti-TNF treatment.

BACKGROUND/AIMS: We aimed to find out if mucosal TNF-α (m-TNF-α) levels have predictive impact on anti-TNF treatment response in Crohn's disease (CD). METHODOLOGY: TNF-alpha values were determined with immunohistochemical staining of intestinal biopsies taken from 35 subjects on anti-TNF treatment for CD and 25 controls. Correlation between m-TNF-α levels and anti-TNF treatment were evaluated. RESULTS: m-TNF-α levels were determined higher in CD group (28.0±8.9 vs 5.8±2.1; P<0.001). m-TNF-α levels were lower in CD patients who were hospitalized (23.0±8.7 vs. 30.3±8.1; P=0.024). Although not to a statistically significant level, favorable parameters such as clinical remission (28.5±8.0 vs. 26.4±11.8, p=0.419) and mucosal healing (29.9±8.2 vs. 23.9±9.2, p=0.097) were increased in patients with high m-TNF-α levels whereas unfavorable parameters such as relapse (26.0±9.4 vs. 29.7±8.2, P=0.107), surgery requirement (22.8±11.1 vs. 29.1±8.1, P=0.147), steroid requirement (25.9±9.0 vs. 28.6±8.9, P=0.595), and anti-TNF intensification (22.3±3.8 vs. 28.5±9.0, P=0.183) were increased in patients with low m-TNF-α levels. CONCLUSIONS: High mucosal TNF-α levels before treatment have favorable effects on anti-TNF treatment response in CD. Presence of high m-TNF-α levels at the diagnosis may be encouraging in early initiation of anti-TNF treatment.

Quantitative evaluation of interstitial fibrosis with Sirius Red in IgA nephritis.

OBJECTIVE: Tubulointerstitial fibrosis is one of the strongest independent predictive factors in determining the prognosis in IgA nephritis. Recently, software-based quantitative measurement of interstitial fibrosis with Sirius Red staining has entered the practice. The objective of this study was to investigate the prognostic value of measurement of interstitial nephritis with this method in IgA nephritis. METHOD: Forty-three patients diagnosed with IgA nephritis with renal biopsy between the years 2005 and 2009 were included in this retrospective observational study. The diagnostic biopsies of 37 patients were examined. Basal data included age, gender, creatinine level, glomerular filtration rate (GFR), presence of proteinuria, hypertension, glomerulosclerosis, mesangial proliferation, and interstitial fibrosis and fibrosis index calculated by the measurement of computed images of Sirius Red positive areas. Final visit included evaluation of development of end-stage renal disease (ESRD), and GFR (whether = 60 mL/min or <60 mL/min). RESULTS: Numbers of patients with hypertension (75% vs. 34.5%; p = 0.050), ESRD development (62.5% vs. 20.7%, p = 0.035), GFR <60 mL/min (87.5% vs. 31%; p = 0.007) were greater; and basal GFR (34.25 ± 25.29 vs. 64.14 ± 35.34; p = 0.048) was lower in high-intensity interstitial fibrosis group (>1000 µm2) compared to low-intensity interstitial fibrosis group (≤1000 µm(2)). CONCLUSION: Quantitative analysis of computed imaging of areas of Sirius Red positive tubulointerstitial fibrosis might serve as an effective novel method to determine the prognosis in IgA nephritis.

The Status of Spermatogenesis in Germ Cell Tumor Bearing Testis and Its Association with Metastatic Disease.

INTRODUCTION: We aimed to evaluate the status of spermatogenesis detected by histological examination of non-tumoral testicular tissues in tumor bearing testis and its association with advanced stage disease. PATIENTS AND METHODS: We retrospectively reviewed patients with testicular germ cell tumors (TGCTs) that undergone radical orchiectomy. All non-tumoral areas of the orchiectomy specimens were examined for the status of spermatogenesis. Patients were divided into two groups as localized (stage I) and metastatic (stage II-III) disease and analyzed separately for seminomatous (SGCT) and nonseminomatous germ cell tumors (NSGCT). RESULTS: Four hundred fifty-four patients were included in our final analysis. Of those, 195 patients had SGCT, and 259 patients had NSGCT. Three hundred and six patients had localized disease at the time of diagnosis. Median (Q1-Q3) age was 31 (26 - 38) years and 102 (22.5%) patients had normal spermatogenesis, 177 (39.0%) patients had hypospermatogenesis and 175 (38.5%) patients had no mature spermatozoa. On multivariate logistic regression analysis, embryonal carcinoma >50% (1.944, 95 %CI 1.054-3.585, P = .033) and spermatogenesis status (2.796 95% CI 1.251-6.250, P = .012 for hypospermatogenesis, and 3.907, 95% CI 1.692-9.021, P = .001 for absence of mature spermatozoa) were independently associated with metastatic NSGCT. However, there was not any variables significantly associated with metastatic SGCT on multivariate logistic regression analysis. CONCLUSION: Our study demonstrated that only 22.5% of patients with TGCTs had normal spermatogenesis in tumor bearing testis. Impaired spermatogenesis (hypospermatogenesis or no mature spermatozoa) and predominant embryonal carcinoma are associated with advanced stage NSGCT.

Effect of the direct renin inhibitor aliskiren in the prevention of experimental contrast-induced nephropathy in the rat.

BACKGROUND: Renal vasoconstriction, activated by the renin-angiotensin system, plays a pivotal role in the pathogenesis of contrast-induced nephropathy (CIN). The purpose of this study was to evaluate the effect of aliskiren, a direct renin inhibitor, for the prophylaxis of experimental CIN in the rat. METHODS: Thirty-two Wistar albino rats were divided into four groups of 8 rats each, namely the control (C), aliskiren (A), contrast media (CM) and aliskiren plus contrast media (ACM) groups. Aliskiren was given orally at a dose of 50 mg/kg/day once daily for 5 consecutive days. CIN was induced by intravenous administration of indomethacin, N-nitro-L-arginine methyl ester and high-osmolar contrast medium meglumine amidotrizoate. Renal function parameters, kidney histology and tubular expression of vascular endothelial growth factor were determined. RESULTS: Mean serum creatinine was significantly lower (p < 0.001) and mean creatinine clearance was higher (p < 0.001) in the ACM group compared with the CM group. However, there were no differences between the ACM and CM groups in terms of tubular necrosis, proteinaceous casts, medullary congestion and vascular endothelial growth factor expression. CONCLUSION: Our preliminary data seem to suggest a potential role of aliskiren for the prophylaxis of CIN in an experimental rat model.

Association of mast cells with microvessel density in urothelial carcinomas of the urinary bladder.

This study aims to investigate the relation of mast cell (MC) accumulation with tumor grade and stage in urothelial carcinomas of the urinary bladder and to determine its relationship with angiogenesis. A total of 78 urothelial carcinomas obtained by transurethral resection were investigated immunohistochemically by using c-Kit (CD117) and anti-CD34. The correlation between MC counts and microvessels was evaluated and compared with histopathologic parameters including tumor stage and grade. There were significant correlations between MC counts, grade, and stage (P < .05; r = 0.69 and 0.63, respectively). However, MC counts in adjacent nontumoral bladder mucosa significantly were higher than the MC counts in tumoral zone (P < .001). On the other hand, significant correlation was found between the number of MCs in tumoral zone and the microvessel density (P < .05, r = 0.56). The results of our study suggest that c-Kit positive MCs in tumoral zone may contribute to tumor angiogenesis and play a significant role in tumor growth and invasion. Further studies are needed to support these observations.

A Rare Case of Xanthomatous Meningioma.

Xanthomatous meningioma is an extremely rare subtype of metaplastic meningiomas with few cases reported in the literature. Histopathologically, it is composed of oval-shaped cells that have central nuclei and cytoplasm with lipid-filled vacuoles, resembling macrophages. Here, the authors present a case of xanthomatous meningioma and discuss the hypotheses related to its origin and the differential diagnosis. A 40-year-old woman presented with an increased headache complaint over the past month. A computed tomography scan revealed a heterogeneous mass on the right parietal lobe, following which a right craniotomy was done. Microscopic examination showed neoplastic meningothelial cells with whorl formation and areas of xanthomatous changes. Both meningothelial and xanthomatous cells were positive with vimentin, progesterone, and epithelial membrane antigen. CD68 and lysozyme were expressed only in the xanthomatous component, and there was no expression with periodic acid-Schiff (PAS) stain and PAS-diastase. As the diagnostic criteria of atypical meningioma were not observed, a diagnosis of xanthomatous meningioma, WHO grade I, was made. Owing to the characteristic xanthomatous changes, its differential diagnosis includes grade II clear cell meningioma, Rosai-Dorfman disease, and hemangioblastoma. To avoid misidentifying these cells as macrophages, a high degree of awareness of this unique subtype of meningioma is required.

Protective effect of the vasopressin agonist terlipressin in a rat model of contrast-induced nephropathy.

BACKGROUND/AIMS: Contrast-induced nephropathy (CIN) remains a leading cause of iatrogenic acute renal failure. Terlipressin, a long-acting analog of vasopressin, may improve renal function. This study aimed to investigate the possible protective effect of terlipressin against the development of experimental CIN in rats. METHODS: Wistar albino rats (n = 32) were allocated randomly into four equal groups of 8 each, i.e. control, terlipressin, contrast media (CM), and terlipressin plus contrast media (TCM). CIN was induced by intravenous administration of indomethacin (10 mg/kg), N-nitro L-arginine methyl ester (L-NAME, 10 mg/kg, twice at 15 and 30 min), and high-osmolar contrast media meglumine amidotrizoate 60%. Renal function parameters, kidney histology, and tubular expression of vascular endothelial growth factor (VEGF) were determined. RESULTS: Mean serum creatinine levels were decreased (p < 0.05) and creatinine clearance (p < 0.05) increased in the TCM group compared with the CM group. Notably, rats in the TCM group displayed less tubular necrosis (p < 0.05), medullary congestion (p < 0.05), and a reduced tubular expression of VEGF (p < 0.05) compared with the CM group. CONCLUSION: These results demonstrate that terlipressin can inhibit the development of CIN.

Focal segmental glomerulosclerosis associated with idiopathic myelofibrosis.

A patient with idiopathic myelofibrosis with nephrotic syndrome is reported. Seven months after the initial diagnosis of myelofibrosis, the patient was presented with dyspnea, generalized edema, heavy proteinuria, massive pleural effusion, and ascites. Renal biopsy showed focal segmental glomerulosclerosis. After starting immunosuppressive therapy consisting of cyclosporine and steroids, his renal function and proteinuria improved and transfusion requirements decreased.

Splenic infarction is not rare in granulomatosis with polyangiitis.

Splenic involvement is rarely reported in patients with ANCA-associated vasculitides (AAVs), particularly in those with granulomatosis with polyangiitis (GPA) and is in fact considered to be underestimated. We aimed to investigate the frequency of splenic lesions-mainly infarction-and related factors in patients with AAVs. Patients with AAV whose abdominal or thoracic computed tomographies (CTs) were performed at or after diagnosis were included in the study. CT images were examined for splenic lesions. Overall, 69 patients (median age at diagnosis 54 [IQR 24] years; 55% with GPA, 29% with microscopic polyangiitis, and 16% with renal-limited disease) were included in the analysis. Splenic pathologies were detected in 19 (28%) patients; 12/19 (63%) splenomegaly and 7/19 (37%) splenic infarction. All patients with splenic infarction exhibited GPA with PR3-ANCA-positive serology. Three of these seven patients had autosplenectomy. Patients with splenic infarction were younger at diagnosis (p = 0.018) with also significantly higher ear-nose-throat (ENT) (%100 vs 37; p = 0.002) and eye involvement (%50 vs %12; p = 0.044) than patients without splenic infarction. Splenic pathologies are not rare in AAVs. Furthermore, infarction can help separate GPA from MPA. In young patients with GPA, particularly those with ENT and eye involvements, physicians should consider splenic infarction.Key Points• Splenic infarction is more common than previously thought in ANCA-associated vasculitides, particularly in granulomatosis with polyangiitis.• Detecting splenic infarction can help differentiate granulomatosis with polyangiitis from other subgroups.

Histopathological changes and tumour necrosis factor-alpha, transforming growth factor-beta and tenascin expression in patients with primary type I membranoproliferative glomerulonephritis in remission.

AIM: Primary type I membranoproliferative glomerulonephritis (MPGN) is a rare cause of glomerular disease with a high relapse rate and poor prognosis. The aim of this study was: (i) to evaluate the histopathological findings associated with remission; and (ii) to document the possible clinical and histopathological factors predicting relapses. METHODS: Eleven type I MPGN patients (five men, six women; mean age, 38.8+/-13.5 years) who were in remission for at least 1 year after the cessation of immunosuppressive drugs were re-biopsied. The intensity of immunostaining for tumor necrosis factor (TNF)-alpha, transforming growth factor (TGF)-beta1, and tenascin was graded from 0 (no staining) to 3+ (maximum staining). RESULTS: Mean baseline mesangial cellularity score and tubulointerstitial infiltration score were reduced and mesangial matrix expansion score was increased at protocol re-biopsies compared to baseline. The glomerular and tubulointerstitial staining scores for TGF-beta1 and tenascin were higher than that of baseline. Reduced tubulointerstitial TNF-alpha expression was found in re-biopsy specimens compared to baseline. Patients have been followed for a mean time of 51.5+/-22.2 months after the protocol biopsy. Eight patients had a relapse. Mesangial cellularity score and glomerular tenascin expression at re-biopsy specimens were higher in relapsed patients compared to those without a relapse. CONCLUSION: Our study shows that mesangial cellularity and tubulointerstitial cell infiltration are reducing whereas mesangial matrix expansion, glomerular and tubulointerstitial TGF-beta1 and tenascin expression are increasing with remission. The higher mesangial cell proliferation and glomerular tenascin scores in remission are associated with the development of relapse.

Tacrolimus rescue therapy in resistant or relapsing cases of primary glomerulonephritis.

BACKGROUND: Patients with resistant or relapsing primary glomerulonephritis (PGN) after conventional immunosuppressive treatment have a worse renal outcome, and treatment of these patients remains problematic. In this study we aimed to determine the effect of tacrolimus in these patients. METHODS: We prospectively studied 15 patients with PGN (3 membranous nephropathy, 6 membranoproliferative GN, 4 focal segmental glomerulosclerosis [FSGS], 1 IgA nephropathy and 1 IgM nephropathy) who were either resistant (n=7) or relapsing (n=8) after or during conventional immunosuppressive therapy. Tacrolimus was started at a dosage of 0.05 mg/kg per day. Prednisolone was either maintained or added to the tacrolimus regimen. RESULTS: The mean duration of tacrolimus treatment was 28.9 +/- 2.4 months. Proteinuria decreased from 6.3 +/- 5.0 g/day to 0.5 +/- 0.6 g/day at the end of follow-up (p=0.001). Complete or partial remission was achieved by 60% and 40% of patients, respectively. Relapse occurred in only 2 patients with FSGS under tacrolimus treatment. There was no significant change in serum creatinine at the end of follow-up (from 1.4 +/- 1.0 mg/dL to 1.2 +/- 0.6 mg/dL, p=0.3). Serum albumin increased from 3.2 +/- 0.5 g/dL to 4.3 +/- 0.3 g/dL (p=0.0001). There were 6 relapses among 10 patients after the withdrawal of tacrolimus. CONCLUSIONS: These data suggest that the combined therapy of tacrolimus and low-dose prednisolone may have a promising role to obtain long-term remission in resistant or relapsing PGN.

Unusual intraconal localization of orbital giant cell angiofibroma.

Giant cell angiofibroma (GCA) is a recently reported rare soft-tissue tumor that can develop in various sites including orbit. Orbital GCAs were mainly located in the eyelid or extraconal regions such as lacrimal gland and conjunctiva. We report an atypical case of a GCA arising in the intraconal area of the orbit in a 65-year-old male patient. The tumor was excised in total by lateral orbitotomy. Histological and immunohistochemical features were consistent with the diagnosis of GCA. No recurrence was observed during the follow-up of over 2 years. GCA is a rare tumor that should be considered in the differential diagnosis of intraconal orbital tumors. Complete surgical removal is the current optimal treatment option.

The blockade of the renin-angiotensin system reverses tacrolimus related cardiovascular toxicity at the histopathological level.

INTRODUCTION: In this study, we investigate the toxic effects of tacrolimus (FK506) on the cardiovascular system at the histopathological level in a rat model and whether these effects can be reversed by the blockade of the renin-angiotensin system (RAS) by either an angiotensin-converting enzyme inhibitor (ACE-inhibitors) or an angiotensin receptor antagonist (ARB). METHODS AND RESULTS: Thirty-one Wistar rats were divided into four groups. FK506 group was treated with FK506 intraperitoneally (i.p.), FK506+ACE-inhibitors and FK506+ARB groups were treated with either quinapril or valsartan orally in addition to FK506. Control group was treated with saline i.p. Histological and immunohistochemical staining of cardiovascular tissue in the FK506 group showed increased vacuolar degeneration (11.2 vs. 5.8, p=0.008), arterial hyalinosis (10.7 vs. 6.3, p=0.036), transforming growth factor-beta (TGF-beta) (12.2 vs. 4.8, p=0.001) and vascular endothelial growth factor expression (VEGF) (10.7 vs. 6.3, p=0.036), elastic van Gieson (11.5 vs. 5.5, p=0.004), and periodic acid Schiff stain scores (12.5 vs. 4.5, p<0.001) compared to the control group. Immunohistochemical scores showed that expression of TGF-beta is up-regulated, and bone morphogenic protein (BMP-7) is down-regulated with FK506 toxicity. Adding RAS blockade with either an ACE-inhibitor or an ARB could reverse FK506 induced changes. Both FK506+ACE-inhibitors and FK506+ARB groups demonstrated decrease in arterial hyalinosis (22.1 vs. 14.4 (FK506+ACE-inhibitor) and 13.6 (FK506+ARB), p=0.09) and vacuolar degeneration (23.1 vs. 16.1 (FK506+ACE-inhibitor) and 12.4 (FK506+ARB), p=0.006) scores compared to the FK506 group. CONCLUSION: Blockade of RAS could reverse the histopathological signs of FK506 induced cardiac toxicity in a rat model.

Extra-Gastrointestinal Stromal Tumor of Prostate.

BACKGROUND: Extra-gastrointestinal stromal tumor is defined as a mesenchymal neoplasm arising from soft tissues outside the gastrointestinal tract. Prostatic extra-gastrointestinal stromal tumor has rarely been noted. CASE REPORT: A 56 year-old man presented with pain in the anal region. A digital rectal examination revealed that the prostate was markedly enlarged with a smooth, bulging surface. Computerized tomography images showed a 6 cm heterogeneous, infiltrative tumor within the prostate gland extending to the trigon of the bladder, left seminal vesicle and rectum. The tru-cut biopsy of the prostate was reported as leiomyoma. It was decided to perform surgery and the masses were easily and completely removed from the adjacent structures. The case was reported as extra-gastrointestinal stromal tumor within the intermediate- risk category with free surgical margins. Four years after the surgery, a locoregional failure was observed and treated with imatinib. CONCLUSION: Stromal tumor, although rare, should be considered in the differential diagnosis in patients with an enlarged prostate.