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Objective: Little is known about the association between whole-blood nicotinamide adenine dinucleotide (NAD + ) levels and nabothian cysts. This study aimed to assess the association between NAD + levels and nabothian cysts in healthy Chinese women., Methods: Multivariate logistic regression analysis was performed to analyze the association between NAD + levels and nabothian cysts., Results: The mean age was 43.0 ± 11.5 years, and the mean level of NAD + was 31.3 ± 5.3 μmol/L. Nabothian cysts occurred in 184 (27.7%) participants, with single and multiple cysts in 100 (15.0%) and 84 (12.6%) participants, respectively. The total nabothian cyst prevalence gradually decreased from 37.4% to 21.6% from Q1 to Q4 of NAD + and the prevalence of single and multiple nabothian cysts also decreased across the NAD + quartiles. As compared with the highest NAD + quartile (≥ 34.4 μmol/L), the adjusted odds ratios with 95% confidence interval of the NAD + Q1 was 1.89 (1.14-3.14) for total nabothian cysts. The risk of total and single nabothian cysts linearly decreased with increasing NAD + levels, while the risk of multiple nabothian cysts decreased more rapidly at NAD + levels of 28.0 to 35.0 μmol/L., Conclusion: Low NAD + levels were associated with an increased risk of total and multiple nabothian cysts., (Copyright © 2024 The Editorial Board of Biomedical and Environmental Sciences. Published by China CDC. All rights reserved.)

Our recent studies indicate that thyroid cysts have clinical implications. Thyroid cysts could have a positive effect on the supply of thyroid hormones. Both hyperthyroidism and hypothyroidism cause hypertension. Hypothyroidism, but not hyperthyroidism, is a risk factor for atherosclerosis. Therefore, thyroid cysts could be associated with hypertension, and atherosclerosis might influence the association between thyroid cysts and hypertension. To evaluate the clinical significance of thyroid cysts, a cross-sectional study was conducted with 1801 Japanese aged 40-74 years. Thyroid cysts were significantly positively associated with hypertension in participants without atherosclerosis. However, there was a significant inverse association in those with atherosclerosis. The potential confounding factor adjusted odd ratios and 95% confidence intervals (95% CIs) were 1.49 (95% CI 1.17-1.90) for participants without atherosclerosis and 0.49 (95% CI 0.24-0.98) for those with atherosclerosis. The present study demonstrates that thyroid cysts have clinical implications because thyroid cysts support thyroid hormone activity. Our findings provide sufficient evidence to develop a risk assessment for hypertension for the general population, even though further research is required.

Anti-thyroid peroxidase antibody (TPO-Ab) is revealed to be inversely associated with thyroid cysts among euthyroid population. TPO-Ab causes autoimmune thyroiditis by bolstering thyroid inflammation. Therefore, at least partly, absence of thyroid cysts could indicate latent thyroid damage. Since participants with subclinical hypothyroidism are reported to have higher HbA1c than normal healthy controls, HbA1c could be inversely associated with thyroid cysts through a mechanism reflecting latent thyroid damage. To investigate the association between HbA1c and thyroid cysts among a euthyroid population, a cross-sectional study was conducted on 1,724 Japanese individuals who were within the normal range of thyroid function [i.e., normal range of free triiodothyronine (T3) and free thyroxine (T4)] and aged 40-74 years. Among this study population, 564 were diagnosed with thyroid cysts. Independently of thyroid related hormones [thyroid stimulating hormone (TSH), free T3, and free T4] and known cardiovascular risk factors, HbA1c was found to be significantly inversely associated with the presence of thyroid cysts. This association remained significant even after this analysis was limited to participants within a normal range of TSH. The fully adjusted odds ratios (ORs) of thyroid cysts for 1 standard deviation (SD) increment of HbA1c were 0.84 (0.74, 0.95) for total participants and 0.80 (0.70, 0.92) for participants within a normal range of TSH. Among participants with normal thyroid function, HbA1c was inversely associated with the presence of thyroid cysts. The absence of thyroid cysts and higher levels of HbA1c could indicate the latent functional damage of the thyroid., Competing Interests: The authors have declared that no competing interests exist.

Background: To evaluate the use of a human fibrin glue (Tisseel) for minor bleeding control and approximation of ovarian defect during transvaginal natural orifice ovarian cystectomy (TNOOC) of benign and non-endometriotic ovarian tumors., Methods: A total of 125 women with benign and non-endometriotic ovarian tumors who underwent TNOOC between May 2011 and January 2020: 54 with the aid of Tisseel and 71 with traditional suture for hemostasis and approximation of ovarian defect. Surgical outcomes such as length of surgery, operative blood loss, postoperative pain score, and postoperative hospital stay were recorded. Before and immediately (10 days) and at 6 months after the procedure, serum anti-Müllerian hormone (AMH) levels were also determined., Results: Complete hemostasis and approximation of ovarian defect were achieved in all cases. No significant difference was noted in the operating time, operative blood loss, postoperative pain scores after 12, 24 and 48 h, length of postoperative stay, and baseline AMH levels between the two groups. The operation did not have a negative effect on the immediate and 6-month postoperative AMH levels in the suture group. However, the decline in the AMH levels was significant immediately after surgery in the Tisseel group, nevertheless, no significant difference was noted in the AMH levels at 6 months (3.3 vs. 1.7 mg/mL; p = 0.042, adjusted p = 0.210)., Conclusion: The use of Tisseel in TNOOC of benign and non-endometriotic ovarian tumors without suturing the ovarian tissue is clinically safe and feasible.

Background: Patients with biliary atresia (BA) with extrahepatic cystic degeneration (BACD) have a unique pathophysiology; however, clinical outcomes and progression of perinatal degeneration are not well-defined. We aimed to investigate the differences in clinical characteristics and outcomes between BACD and isolated BA (IBA)., Methods: We performed a retrospective analysis of patients with BA who underwent Kasai portoenterostomy (KPE) from August 1997 to January 2018 and compared the clinical features and outcomes between BACD (n = 21) and IBA (n = 237). Matched-pair analysis for age and sex was performed between BACD and IBA groups to reduce confounding., Results: Before matched-pair analysis, we found that BACD patients were younger at KPE (45 vs. 64 days, p = 0.008), showed lower total bilirubin at the 3-month follow-up (0.5 vs. 1.4 mg/dL, p = 0.002), and higher 5-year native liver survival rate (95.2% vs. 61.4%, p = 0.006) than IBA patients. After matching, the BACD group showed significantly lower total bilirubin levels at the 3-month follow-up (0.5 vs. 1.5 mg/dL, p = 0.036) and higher 5-year native liver survival rate (95.2% vs. 57.5%, p = 0.006) than the IBA group., Conclusion: BACD demonstrated higher bilirubin clearance and native liver survival rates than IBA., Levels of Evidence: Treatment Study, Level III., (Copyright © 2020 Elsevier Inc. All rights reserved.)

Previous studies have suggested that pathogenic variants in interferon regulatoryse factor 6 (IRF6) can account for almost 70% of familial Van der Woude Syndrome (VWS) cases. However, gene modifiers that account for the phenotypic variability of IRF6 in the context of VWS remain poorly characterized. The aim of this study was to report a family with VWS with variable expressivity and to identify the genetic cause. A 4‑month‑old boy initially presented with cleft palate and bilateral lower lip pits. Examination of his family history identified similar, albeit milder, clinical features in another four family members, including bilateral lower lip pits and/or hypodontia. Peripheral blood samples of eight members in this three‑generation family were subsequently collected, and whole‑exome sequencing was performed to detect pathogenic variants. A heterozygous missense IRF6 variant with a c.1198C>T change in exon 9 (resulting in an R400W change at the amino acid level) was detected in five affected subjects, but not in the other three unaffected subjects. Moreover, subsequent structural analysis was indicative of damaged stability to the structure in the mutant IRF protein. Whole‑transcriptome sequencing, expression analysis and Gene Ontology enrichment analysis were conducted on two groups of patients with phenotypic diversity from the same family. These analyses identified significant differentially expressed genes and enriched pathways in these two groups. Altogether, these findings provide insight into the mechanism underlying the variable expressivity of VWS.

Although carbohydrate antigen 19-9 (CA 19-9) may be elevated in benign diseases, elevated CA 19-9 may cause a fear of cancer and unnecessary follow-up studies. Research on how to approach systematically in this case is very limited. The purpose of this study was to analyze the clinical features and the causes of CA 19-9 elevation without evidence of malignant or pancreatobiliary diseases. We retrospectively reviewed the medical records of patients who had CA 19-9 elevation (≥80 U/mL) and were found to be unrelated to cancer after follow-up. After exclusion, 192 patients were included in this study. The median level of CA 19-9 was 136.5 U/mL. The causes of CA 19-9 elevation were determined in 147 (76.6%) patients, and that was unknown in 45 (23.4%). The estimated causative diseases were hepatic diseases in 63 patients, pulmonary diseases in 32, gynecologic diseases in 38, endocrine diseases in 13, and spleen disease in 1. Of 45 patients with unknown cause, 35 had normalization of CA 19-9 and 10 had persistently elevated CA 19-9. In conclusion, CA 19-9 elevation without malignancies or pancreatobiliary diseases should be systematically evaluated and followed up. We suggest an algorithm to investigate the causes and follow up these patients.

Objective: Primary objective is to identify risk factors of endometriotic-cyst associated ovarian cancer (EAOC). Secondary objective is to evaluate the clinical characteristics of EAOC patients., Materials and Methods: A retrospective case-control study was conducted by analyzing data of patients from 1999 to 2014. Cases were endometriotic-cyst associated ovarian cancer with pathologically confirmed diagnosis. Controls were randomly selected with year-matched patients with benign ovarian endometriotic cyst. Univariate and multivariate with logistic regression analyses were used to identify patients' characteristics that were risk factors for endometriotic-cyst associated ovarian cancer., Results: Altogether, 158 controls and 79 EAOC cases were recruited. Mean age of the EAOC group was 13 years older than that of the control group (49 vs. 36 years). The most common stage of EAOC was stage I (59.74%). Clear cell subtype is the most commonly found in this population (60.76%). Univariate analysis showed that age ≥42 years, menopause, weight loss, cyst diameter ≥8.33 cm, presence of solid area, bilaterality and CA 125 higher than 117.6 units/ml were significant. Multivariate analysis showed that patients with age ≥42 years (OR 7.69, 95%CI: 2.47, 23.87), menopause (OR 33.19, 95%CI: 2.37, 465.12), weight loss (OR 11.94, 95%CI: 1.52, 94.08), cyst diameter ≥ 8.3 cm (OR 10.56, 95%CI: 4.39, 25.35) and presence of solid area by ultrasonography (OR 6.70, 95%CI: 2.19, 22.35) were significant risk factors for EAOC., Conclusion: Advanced age, menopause, weight loss, cyst diameter ≥ 8.33 cm and presence of solid area from ultrasonography were important risk factors for EAOC., Competing Interests: Declaration of Competing Interest The authors declare no conflict of interest., (Copyright © 2020. Published by Elsevier B.V.)

Background: To determine the role of E-selectin gene S128R polymorphism on the enlargement of renal cysts in patients with polycystic kidney disease (PKD)., Materials and Methods: 76 PKD patients with no comorbidity were enrolled in the study. Serum E-selectin levels were analyzed by enzyme-linked immunoabsorbent assay (ELISA). E-selectin gene S128R (561 A>C, rs: 5361) polymorphism was examined by polymerase chain reaction restriction fragment length (PCR-RFLP). Magnetic resonance imaging was performed at baseline evaluation and at the end of the 1 st year to determine cyst enlargement and total kidney volume (TKV)., Results: No significant difference was identified between AA genotype and AC or CC variants of E-selectin gene S128R polymorphism in terms of age, disease duration, baseline cyst volume, cyst volume at the 12 th month, baseline dominant cyst volume, and dominant cyst volume at the 12 th month. In contrast, a significant difference was determined between the groups with regard to the change of TKV (2.9 ± 13.4 vs. 5.2 ± 16.3 mm 3 ; respectively, p = 0.01). In the correlation analysis, the serum E-selectin level was significantly correlated to glucose, alanine transaminase, creatinine, calcium, phosphorus, total protein, albumin, and end diastolic volume (p = 0.0001, p = 0.001, p = 0.03, p = 0.021, p = 0.023, p = 0.002, p = 0.003, and p = 0.047, respectively). Multivariate logistic regression analysis demonstrated a 1.32-fold higher risk of cyst enlargement in patients with CC polymorphism when compared to AA genotype (p = 0.052), but not between AA and AC genotypes or CC and AC genotypes., Conclusion: PKD patients with CC variants of the E-selectin gene S128R polymorphism are at greater risk of cyst enlargement. The results of the present study should be confirmed with further studies with large sample size and longer duration of follow-up.

CA125/MUC16 is an ovarian tumor cell marker widely used as a biomarker in epithelial ovarian carcinoma. CA125/MUC16 is also used for evaluation of the ROMA (Risk of Ovarian Malignancy Algorithm) value. In this work, a Surface Plasmon Resonance Imaging (SPRI) biosensor for circulating CA125/MUC16 has been developed. The anti-MUC16 antibody was attached to a gold chip via a cysteamine linker. The EDS/NHS protocol was used for the covalent attachment of the antibody. The developed biosensor is specific for CA125/MUC16, and exhibits good recovery and acceptable precision. Its linear response range (2.2-150 U/ml) is well suited to determination of the marker in the blood serum of a healthy control group and, after appropriate dilution, of patients with ovarian cancer. CA125/MUC16 was determined in two series of real samples: blood serum from patients with ovarian cancer and endometrial cysts. The method was validated by parallel determination of the samples using the chemiluminescent Architect i2000 method., (Copyright © 2019 Elsevier B.V. All rights reserved.)

This study was undertaken to evaluate the correlation between preoperative Serum markers and pelvic adhesions in endometriosis patients and to explore the markers' clinical value for outcome prediction. Preoperative blood Serum and CA 125 results were obtained and pelvic adhesion scores were calculated. The patient group with adhesion scores less than 28 points was defined as the mild adhesion group, and those with a score of 28 or more were members of the severe adhesion group. The CA 125 level was significantly higher in the severe adhesion group than in the mild adhesion group. The CA 125 level, size of the largest cyst, and WBC count were associated with the level of pelvic adhesion. Adhesion scores were significantly higher in the CA 125 ≥ 35 U/mL group than in the CA 125 < 35 U/mL group. Patients with a preoperative CA 125 level higher than 35 U/mL are at high risk for pelvic adhesion., (Copyright © 2019 Elsevier Masson SAS. All rights reserved.)

A seizure is a symptom of brain dysfunction, resulting in 1.6 million emergency department visits each year. The evaluation of new seizures in the emergency department is a process looking for triggers of seizures such as toxins, mass lesions, or metabolic derangements. Maintaining a broad differential diagnosis is essential to prevent premature closure of the diagnostic evaluation. Timing of neurologic imaging and electroencephalogram depends on the clinical situation. In this case, a young woman presents with a "new-onset" seizure with subsequent elevation of white blood cells and serum lactate levels. Neurologic imaging identified a large dermoid cyst in the frontal lobe. We review how seizures can elevate the serum lactate distinguishing a general tonic-clonic seizure from pseudoseizures and how dermoid cysts can be implicated in seizures.

The objectives of this study were to investigate the relationship between cerebrospinal fluid lactate and serum concentrations in dogs with clinical signs of central nervous system disease and to establish if cerebrospinal fluid lactate (CSF) concentrations are higher in dogs with structural intracranial disease (Group Pos-MRI) compared to dogs that have clinical signs of intracranial disease but no structural brain disease (Group Neg-MRI) based on magnetic resonance imaging (MRI) findings. Using a prospective study canine blood and cerebrospinal fluid were collected in 24 dogs with neurological signs after undergoing brain MRI. Dogs were divided in 2 groups. No significant difference between serum lactate (1.57 ± 0.9 mmol/L) and CSF lactate concentration (1.34 ± 0.3 mmol/L) was detected. There was a direct correlation between CSF and serum lactate concentration ( R = 0.731; P = 0.01). No significant difference was found in CSF lactate concentration between the 2 groups of dogs ( P = 0.13).

Aim: Liver cysts are the most common extrarenal manifestation in patients with autosomal dominant polycystic kidney disease (ADPKD). Carbohydrate antigen 19-9 (CA19-9) is generally used as a marker for biliopancreatic malignancies, although CA19-9 levels in patients with ADPKD are largely unknown., Methods: A prospective observational study of 53 ADPKD patients and 83 non-ADPKD control subjects was performed. The serum levels of CA19-9 were studied to evaluate the association with clinical parameters and liver cysts., Results: The serum CA19-9 levels were significantly higher in the ADPKD group than in the control group (32.9 U/mL vs. 9.8 U/mL, respectively, P < 0.001). The serum CA19-9 levels in the ADPKD group were positively correlated with the mean blood pressure (rho = 0.335, P < 0.05), gamma-glutamyl transferase (GTP) levels (rho = 0.541, P < 0.001), the largest cyst size (rho = 0.536, P < 0.001) and the liver cyst volume (rho = 0.682, P < 0.001). Multiple regression analyses showed that the gamma-GTP levels (P < 0.001) and the liver cyst volumes (P < 0.001) were independent predictors for serum CA19-9 levels., Conclusions: Serum CA19-9 levels are significantly elevated and appear to be dependent on the gamma-GTP levels and the volume of liver cysts in patients with ADPKD. Our findings indicate that the measurement of the baseline CA19-9 level in each patient with ADPKD may be useful for the interpretation of the value and the differential diagnosis of liver diseases, particularly the liver cyst infection., (© 2016 Asian Pacific Society of Nephrology.)

Background: Parathyroid cysts are relatively uncommon lesions and are often misdiagnosed. We evaluate our experience in the diagnosis of and therapy to correct parathyroid cystic lesions., Methods: We retrospectively reviewed a series of 32 patients with parathyroid cysts who were admitted to our department between July 2011 and November 2016. Clinical pathological features of the patients, including age, gender, location, size, ultrasonography, histopathology, surgery, and follow-up, were analyzed., Results: There were 22 female and 10 male participants with a median age of 46.7 years old (27-76 years old). Only two cysts were found in the superior mediastinum. The rest were located under the lower pole of the thyroid. All of the patients underwent ultrasonography scans and serum parathyroid hormone (PTH) assays. Three patients had elevated serum PTH levels, and they were further scanned with Tc99m sestamibi as functional cysts. In 29 cases of nonfunctional cysts, 3 cases were preoperatively diagnosed by cystic aspiration with PTH detection. The rest were diagnosed by postoperative immunopathology. All of the patients underwent cystectomy, and 24 patients also underwent thyroidectomy. There was a significant difference in cyst diameter size between the cystectomy alone and cystectomy with thyroidectomy groups (4.0 ± 2.0 vs 1.5 ± 1.0 cm; p < 0.05). No participant experienced recurrence during the median 36 months of follow-up., Conclusions: Cystic lesions located under the lower pole of the thyroid gland should be considered to have originated at the parathyroid gland. Cystic aspiration with PTH detection or postoperative immunopathology can lead to a definitive diagnosis. Cystectomy is still a commonly used and effective treatment.

Introduction: Tailgut cyst are congenital cystic lesion arising from remnant of the embryological postnatal gut. Tailgut cyst are multinodular, uncapsulated and usually well-circumscribed. Presacral cysts are rare in adult and most of the lesions are benign. Malignant degeneration can occur, however is extremely rare., Case Report: We present the case of a 74 years old woman with slow increase in size and malignant degeneration of a tailgut cyst. Five years before, during the follow up after mastectomy for cancer, she manifested rise of CA 19-9 tumor marker and a presacral cystic collection on thoraco-abdominal CT. She was followed with CT and MRI that showed that the cyst, with a solid component of the wall, was growing larger. After a five-year evolution, the cyst was resected. The histological examination on the solid component demonstrated intestinal adenocarcinoma., Conclusion: MRI ant TC can play essential role in the preoperative detection and characterization for the differential diagnosis, treatment strategies and evaluate neoplastic degeneration. Due to the risk of malignancy surgical resection must be performed after the diagnosis. Surgical therapy is mandatory when the cyst grow larger and a solid component is present., Key Words: Presacral cyst, Retrorectal tumors, Tailgut cyst.

A pineal cyst is a benign affection of the human pineal gland on the borderline between pathology and normality. Only a small percentage of patients present with symptoms and a surgical treatment is indicated in highly selected cases. A melatonin secretion in patients with a pineal cyst before and after a pineal cyst resection has not been studied yet and the effect of surgery on human metabolism is unknown. The present study examined melatonin, cortisol and blood glucose secretion profiles perioperatively in a surgical group of 4 patients. The control group was represented by 3 asymptomatic patients with a pineal cyst. For each patient, 24-h circadian secretion curves of melatonin, cortisol and glycemia were acquired. An analysis of melatonin profiles showed an expected diurnal pattern with the night peak in patients before the surgery and in the control group. In contrast, melatonin levels in patients after the surgery were at their minimum throughout the whole 24-h period. The cortisol secretion was substantially increased in patients after the surgery. Blood glucose sampling showed no statistically significant differences. Clinical results demonstrated statistically significant headache relief measured by Visual Analogue Scale in patients after the surgery. Despite the small number of examined patients, we can conclude that patients with a pineal cyst preserved the physiological secretion of the hormone melatonin while patients who underwent the pineal cyst resection experienced a loss of endogenous pineal melatonin production, which equated with pinealectomy. Surprisingly, cortisol secretion substantially increased in patients after the surgery., (Copyright © 2017 Elsevier Ltd. All rights reserved.)

Background: The aim of this study was to investigate the incidence of benign histology after partial nephrectomy (PN) in patients with presumed malignancy from preoperative imaging. Furthermore, preoperative predictors of benign lesions and perioperative outcomes were also assessed., Methods: A series of patients undergoing PN for renal masses was identified using a prospectively maintained database. Patients were excluded for known genetic conditions, if more than one renal mass was resected, or if standard preoperative imaging was not suspicious for renal-cell carcinoma (RCC). Differences in characteristics between patients with benign and malignant pathology were assessed., Results: A total of 916 patients were identified who underwent PN between 2007 and 2015, including 129 (14.1%) patients with a final diagnosis of benign disease. The most common types of benign pathology were oncocytoma (n = 66, 51.2%), angiomyolipoma (n = 37, 28.7%), and complex cysts (n = 10, 7.8%). Low body mass index (BMI) [0.96 (0.92-0.99) p = 0.02], low R.E.N.A.L. score [0.86 (0.76-0.96) p = 0.007], and low preoperative creatinine [0.37 (0.14-0.91) p = 0.04] predicted benign histology in multivariate analysis. Tumor size was a significant predictor in additional modeling [0.81 (0.69-0.94) p = 0.008]. Patients with benign histology had significantly shorter operative times (p < 0.001) and less estimated blood loss (p < 0.001), and there was no difference in complication (p = 0.93) or blood transfusion (0.24) rates., Conclusions: In this study, the rate of benign pathology after PN for presumed RCC is 14.1%. BMI, R.E.N.A.L. score, and preoperative creatinine are predictive of benign histology, but the ability of different variables to predict benign lesions may be influenced by the distribution of benign tumor subtypes, reflecting potential unidentified selection bias.

Context: Cystic prolactinomas are considered resistant to volume reduction by dopamine agonists (DAs). Although several individual case reports and small case series have suggested that DAs may reduce these lesions, larger series using standardized imaging metrics are lacking., Objective: The objectives of the study were to assess the efficacy of DAs on cyst size in patients with predominantly cystic prolactinomas and to characterize the clinical course and treatment outcomes in these patients., Design: This study was a retrospective review., Setting: The study was conducted at a tertiary referral center., Subjects: The study comprised 30 adults with cystic prolactinomas treated at Massachusetts General Hospital., Intervention(s): The interventions included treatment with Das and transsphenoidal surgery., Main Outcome Measure(s): Cyst volume calculated by a commercial software and pituitary hormone function were measured., Results: Median age was 31.5 years (interquartile range [IR] 24.5-39.2), and 24 of 30 were female. Median length of follow-up was 3.05 years (IR 1.04-5.28). Twenty-three of 30 patients received initial treatment with DAs. Median cyst volume for these patients was 435 mm 3 (IR 255-1785). Persistent cyst reduction occurred in 20 of 22 patients after DA treatment. Median cyst volume reduction was 83.5% (IR 48.8-96.2). Median time to cyst reduction was 24.6 weeks (range 2.6-73). Chiasm compression resolved in four of five patients, and nongonadal anterior hypopituitarism improved in five of six. Transsphenoidal surgery was ultimately performed in 15 of 30 patients., Conclusions: Significant cyst reduction occurred in the majority of patients treated with DAs, including those with larger lesions and chiasm compression. This study is the first formal analysis of cyst reduction with DAs in patients with cystic prolactinomas, and contrary to long-held assumptions, our results suggest that medical therapy may be effective in many such patients.

Background & Aims: Somatostatin analogues reduce liver volumes in polycystic liver disease. However, patients show considerable variability in treatment responses. Our aim was to identify specific patient, disease or treatment characteristics that predict response in polycystic liver disease during somatostatin analogue therapy., Methods: We pooled the individual patient data of four trials that evaluated long-acting somatostatin analogues (120 mg lanreotide or 40 mg octreotide) for 6-12 months in polycystic liver disease patients. We performed uni- and multivariate linear regression analysis with preselected patient, disease and drug variables to identify independent predictors of response, defined as per cent change in liver or kidney volume (in ADPKD subgroup). All analyses were adjusted for baseline liver volume and centre., Results: We included 153 polycystic liver disease patients (86% female, median liver volume 4974 ml) from three international centres, all treated with octreotide (n = 70) or lanreotide (n = 83). Mean reduction in liver volume was 4.4% (range -31.6 to +9.4%). Multivariate linear regression revealed that elevated baseline alkaline phosphatase was associated with increased liver volume reduction during therapy (-2.7%, 95% CI -5.1 to -0.2%, P = 0.04), independently of baseline liver volume. Somatostatin analogue type, underlying diagnosis and eGFR did not affect response. In our ADPKD subpopulation (n = 100), elevated alkaline phosphatase predicted liver volume reduction (-3.2%, P = 0.03) but did not predict kidney volume reduction (+0.1%, P = 0.97). Total gastro-intestinal symptom severity decreased with therapy in a subgroup analysis (n = 95; P < 0.001)., Conclusion: Alkaline phosphatase is a liver-specific, independent predictor of response in polycystic liver disease during somatostatin analogue therapy., (© 2015 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

Patients with autosomal dominant polycystic kidney disease and polycystic liver disease (PLD) often have elevated serum levels of alkaline phosphatase (ALP) and gamma-glutamyl transpeptidase (GGT). Ursodeoxycholic acid (UDCA) is used to treat biliary tract diseases, but its effect on PLD remains unclear. UDCA was administered for 1 year at a dose of 300 mg daily to seven PLD patients with elevated ALP or GGT levels who were selected for this treatment by experienced clinicians. Laboratory data and liver volumes were compared among three time points: 1 year before UDCA treatment, at the start of UDCA therapy, and 1 year after the start of therapy. Median GGT did not show a significant change between 1 year before UDCA (180 IU/L) and the start of UDCA therapy (209 IU/L), but it decreased significantly to 98 IU/L after 1 year of UDCA therapy (P = 0.015 vs. the start of therapy). ALP showed a significant increase from 1 year before UDCA (456 IU/L) to the start of UDCA therapy (561 IU/L), and then decreased significantly after 1 year of UDCA therapy (364 IU/L). Median liver volume did not show any significant changes among these three time points of assessment. UDCA may be effective for reducing biliary enzyme levels and inhibiting the growth of liver cysts in patients with PLD., (© 2015 International Society for Apheresis, Japanese Society for Apheresis, and Japanese Society for Dialysis Therapy.)

We report a case of a patient who presented with an extremely high serum prostate specific antigen (PSA) level and underwent radical prostatectomy for presumed prostate cancer. Surprisingly, the whole mount prostatectomy specimen showed only small volume, organ-confined prostate adenocarcinoma and a large, benign intraprostatic cyst, which was thought to be responsible for the PSA elevation., (2016 BMJ Publishing Group Ltd.)

Background: Ciliated hepatic foregut cyst (CHFC) is a rare cystic lesion most commonly identified in segment 4 of the liver that arises from the embryonic foregut. The classic histologic pattern is comprised of 4 distinct layers (inner ciliated epithelial lining, smooth muscle, loose connective tissue, fibrous capsule). Although rare, cases of metaplastic and malignant epithelial lining have been described in CHFC., Methods: We report 6 additional cases of CHFC, one of which had gastric metaplasia of the cyst lining, and review all reported cases of CHFC in the English literature. We describe the clinicopathologic analysis of 6 cases, with selective immunohistochemical analysis on 1 case with gastric metaplasia., Results: Cases occurred in 4 women and 2 men (average age 55 years, range 42 to 67 years). Cysts ranged in size from 0.7 to 17 cm (average 7.2 cm) and were grossly tan-pink to white with blood-filled contents. The majority were located in segment 4 of the liver, however 2 were located in the porta hepatis. Tumor serologies (CA19-9 and/or CEA) were performed in 3 cases; 1 case demonstrated elevated CA19-9, and 2 cases had laboratory values within normal limits. All cases showed the classic histologic findings, however one case additionally had extensive gastric metaplasia., Conclusions: In conclusion, CHFC is a rare diagnostic entity that should be considered in the differential diagnosis for cystic hepatic lesions, particularly those located in segment 4 of the liver. Metaplasia and squamous carcinoma can occur, therefore complete surgical excision is the recommended treatment.

Purpose: Although computed tomography (CT) has been used previously to assess disease severity in lymphangioleiomyomatosis (LAM), the associations between the extent of pulmonary cysts on CT and six-minute walk test (6MWT), matrix metalloproteinases (MMPs) and vascular endothelial growth factor (VEGF-D) are not well established. We performed a cross-sectional study to quantify the extent of pulmonary cysts in CT and to establish their correlations with pulmonary function tests (PFTs), 6MWT results, including a composite index (desaturation-distance ratio, DDR), and levels of VEGF-D and MMPs in LAM., Methods: Twenty-three LAM patients underwent CT scanning to automatically quantify the extent of pulmonary cysts and performed PFTs and 6MWT. Serum levels of MMP-2, MMP-9, and VEGF-D were also measured., Results: The severity of pulmonary cystic involvement was mild (the extent of cysts was 6.8 %) and correlated best with FEV1/FVC (r = -0.84), residual volume (r = 0.66), DLCO (r = -0.82), the DDR index (r = 0.77), and desaturation during the 6MWT (r = -0.81). There was a weak correlation with VEGF-D (r = 0.45), but no association was found with MMP-2 and MMP-9., Conclusions: The severity of pulmonary cystic involvement was mild in this sample of LAM patients and correlated best with airway obstruction, air trapping, reduced DLCO, the DDR index, and desaturation during the 6MWT. Serum VEGF-D cannot be completely defined as a valuable marker of disease severity and there may be a mechanism independent of MMPs to explain the formation of pulmonary cysts.

History and Admission Findings: A 55-year-old woman presented comfortable and well-nourished for a check up. The physical examination remained without pathological results. However lipase parameter in serum was conspicuously elevated we performed an ultrasound examination., Examinations: An abdomen sonography revealed besides regular pancreas morphology a hypoechoic lesion of 11 × 10 mm in the liver and multiple hypoechoic inhomgeneous lesions in the spleen. Because of the inhomogeneous results and septa we decided to arrange an abdomen computer tomography (CT) in which the complex of cysts was stated with a size of 59 × 44 mm. As the differential diagnosis of the cysts could be an echinococcosis or amoebic infection we performed a serological analysis at the first appointment and 4 weeks later. Both test results were negative., Treatment and Course: Eight years later we indicated a magnetic resonance tomography (MRI with MRCP) to exclude a compression of the pancreatic duct by the splenic or liver cysts because the lipase und C-reactive protein (CRP) in serum was rising up again . The MRI scan showed a progression of the splenic cysts complex to 88 × 69 mm and the liver cyst to 25 × 20 mm without signs for compression or a malignant process. A correlation between elevated lipase and splenic cysts was not evident. Despite progression of the splenic cysts we decided in view of the comfortable condition of the patient and negative serological analysis for a conservative proceeding in terms of regular check ups., Conclusion: From our point of view also in extended non-parasitic splenic cysts it is reasonable to follow a nonoperative regime as long as clinical complaints, parasitic genesis and malignant process can be excluded., (© Georg Thieme Verlag KG Stuttgart · New York.)

Objective: To investigate the clinical manifestation, diagnosis, treatment of parathyroid occupying lesions., Method: The clinical data of 26 patients with parathyroid occupying lesions between January 2003 and October 2012 at Dalian Central Hospital were retrospectively analyzed., Result: There were 2 cases of parathyroid cancer, 11 cases of parathyroid adenoma, 13 cases of parathyroid cysts in this study. All the cases,except 13 cases of cysts, are accompanied by symptoms of hyperparathyroidism. The symptoms were as follows:26 cases of neck lump, 11 cases of osteoporosis/osteitis fibrosa cystica,9 cases of urinary symptom, 8 cases of digestive symptom. Serum calciumion level and serum parathyroid hormone(PTH) level were examined qualitatively before operation. Ultrasonography, CT, MRI were used in diagnosing and locating parathyroid occupying lesion before operation. 2 cases of parathyroid cancer, 11 cases of parathyroid adenoma and 13 cases of parathyroid cysts with preoperative and postoperative diagnosis coincidence rate was 0 (0/2), 91% (10/11), 69% (9/13). All patients were treated with operation. 2 cases of parathyroid cancer survived with out recurrence during follow up for 17 months and 27 months after operation. 11 cases of parathyroid adenoma and 13 cases of parathyroid cysts were treated with operation,achieved complete remission and the outcome was no recurrence., Conclusion: There lack of specific clinical symptoms of parathyroid occupying lesions,neck lump are common manifestations. Examination of serum calcium and PTH level together with ultrasonography, CT, MRI, 99mTc-MIBI is helpful to diagnose parathyroid occupying lesions. Parathyroid cancer preoperative diagnosis is difficult . Parathyroid occupying lesions determine the diagnosis depends on pathology. Surgery should be done as primary treatment.

Unlabelled: Cancer antigen CA-125 is a marker that is primarily used to differentiate benign from malignant tumors as well as to monitor response to ovarian cancer treatment. Taken as a separate marker, it displays low sensitivity and specificity in ovarian cancer diagnosis; however, in combination with other markers it may be successfully applied especially in postmenopausal women. Elevated CA-125 levels in blood serum indicate cancerous as well as non-cancerous diseases. Research aiming to determine environmental factors that may have influence on antigen CA-125 level, and thus on the assessment of this marker's application in gynecological and oncological diseases continues., Objectives: the aim of the present research is an attempt to estimate the influence of nicotinism on antigen CA-125 in blood serum in patients with diagnosed benign ovarian tumors including endometrial cysts., Material and Methods: 174 women aged 16-85 years with diagnosed benign ovarian tumor were qualified for the study. In all patients level of antigen CA-125 in blood serum was assessed preoperatively and nicotinism history was taken. Also transvaginal ultrasound was performed to obtain preliminary diagnosis. Smoking and non-smoking patients were classified into two groups, namely of those with histopathologically confirmed cysts of endometrial type and those with non-endometrial benign ovarian tumors., Results: statistical analysis did not prove any dependence between the CS-125 antigen level and nicotinism in any of these groups. Also additional analysis with division into premenopausal and postmenopausal patients did not determine any statistically significant dependence., Conclusion: Nicotinism does not significantly influence the CA-125 antigen level in patients with benign However, the connection between the addiction severity and its influence on antigen CA-125 in blood serum cannot be excluded. ovarian tumors or endometrial cysts.

SPECT/CT imaging can provide improved diagnostic information over traditional planar scintigraphy and SPECT, with more precise anatomic localization and observer confidence. We present 2 patients with polycystic liver disease (PCLD), both with constitutional symptoms and bacteremia. In WBC SPECT/CT images revealed increased WBC localization within a single liver cyst in each case, subsequently drained under imaging guidance. Cultures confirmed the presence of infection, allowing for appropriately directed antibiotic therapy and successful treatment outcomes. These cases illustrate the incremental value of In WBC SPECT/CT fusion imaging for the evaluation of bacteremia in complicated patients.

Hepatic cysts are increasingly found as a mere coincidence on abdominal imaging techniques, such as ultrasonography (USG), computed tomography (CT) and magnetic resonance imaging (MRI). These cysts often present a diagnostic challenge. Therefore, we performed a review of the recent literature and developed an evidence-based diagnostic algorithm to guide clinicians in characterising these lesions. Simple cysts are the most common cystic liver disease, and diagnosis is based on typical USG characteristics. Serodiagnostic tests and microbubble contrast-enhanced ultrasound (CEUS) are invaluable in differentiating complicated cysts, echinococcosis and cystadenoma/cystadenocarcinoma when USG, CT and MRI show ambiguous findings. Therefore, serodiagnostic tests and CEUS reduce the need for invasive procedures. Polycystic liver disease (PLD) is arbitrarily defined as the presence of > 20 liver cysts and can present as two distinct genetic disorders: autosomal dominant polycystic kidney disease (ADPKD) and autosomal dominant polycystic liver disease (PCLD). Although genetic testing for ADPKD and PCLD is possible, it is rarely performed because it does not affect the therapeutic management of PLD. USG screening of the liver and both kidneys combined with extensive family history taking are the cornerstone of diagnostic decision making in PLD. In conclusion, an amalgamation of these recent advances results in a diagnostic algorithm that facilitates evidence-based clinical decision making.

Blood-filled cysts of larger size attached to the heart valves represent a very rare finding in adults. We report here a case of a blood-filled cyst attached to the papillary muscle, demonstrating the importance of multimodal preoperative diagnostic imaging combining both echocardiography and magnetic resonance imaging.

The cyclic nature of some of headache disorders is closely related to melatonin, which is secreted by the pineal gland. We report a 29-year-old male patient with a 2.5-year history of headaches that woke him in the middle of the night. These headaches were pulsatile and continued until sunrise. During these attacks he also suffered from allodynia over the scalp, bilateral conjunctival hyperemia, and nervousness. His brain MRI showed a 5mm by 4mm neuroepithelial cyst in the pineal gland. The peak plasma melatonin level that was measured at 2 am was 28 pg/mL. The patient underwent oral melatonin treatment (6 mg/day). After 1 month he experienced a 70% reduction in his symptoms. When the melatonin dosage was increased to 10mg/day he became headache-free, and 5 months after the treatment began, had no complaints. His 5-month follow-up plasma melatonin level at 2 am was 61 pg/mL. To our knowledge this is the first report of a patient with nocturnal headache associated with a low level of melatonin due to a neuroepithelial cyst in the pineal gland., (Copyright © 2011 Elsevier Ltd. All rights reserved.)

Blood cysts of the mitral valve are mostly benign diverticuli lined by endothelium and filled with blood and can be safely monitored with echocardiographic follow-up. We report a case of asymptomatic blood cyst of the mitral valve in a 63-year-old woman referred for a systolic murmur. At 3-year echo follow-up, the patient is free from notable clinical events.

An epithelial cyst in an intrapancreatic accessory spleen (ECIAS) is a rare disease that is commonly misdiagnosed preoperatively. To identify the clinical and imaging features of ECIAS, we reviewed the relevant medical literature. Twenty-one cases of ECIAS were identified, including our own. The cases were mainly diagnosed as mucinous cystic neoplasm (MCN) preoperatively based on clinical and imaging features, such as, a woman in middle age; elevation of serum CA19-9 levels; location in the tail of the pancreas; and a solid component resembling a mural nodule. ECIAS is another lesion to be considered in the differential diagnosis of MCN.

Polycystic thyroid disease (PCTD) is characterized by multiple thyroid cysts detected by ultrasonography, the absence of thyroid autoantibodies, and susceptibility to the development of hypothyroidism due to a high iodine intake. It is necessary to obtain histopathological information on PCTD in order to clarify the cause of hypothyroidism. We retrospectively reviewed three patients with PCTD and small papillary thyroid cancer who underwent thyroidectomy. We observed the thyroid tissues pathologically in areas with and without multiple cysts, and compared them with those of multinodular goiter with cysts. In the patients with PCTD, there were multiple enlarged follicles that resembled enlarged normal follicles and differed from those found in multinodular goiter in terms of their shape. Huge follicles corresponded to the cysts that were detected by ultrasonography. Each follicle contained colloid. Follicular cells in enlarged follicles comprised low cuboidal epithelium that appeared normal. These findings were common in the 3 patients with PCTD. In Conclusion the PCTD patients had multiple enlarged follicles that seemed to decrease the total number of follicular cells, and may be a cause of hypothyroidism. We believe that PCTD is a new entity of thyroid disease based on the pathological findings.

Epithelial splenic cysts are rare entities which surgeons may not have previously encountered. Their management is based on historical studies. A 21-year-old woman presented with a cyst of the spleen that produced a high serum concentration of carbohydrate antigen CA 19-9. A partial splenectomy with removal of the entire cyst and preservation of the splenic parenchyma by laparotomy was performed and the patient made an uneventful recovery. A microscopic rupture of the wall of the cyst and blood degradation products in the fluid of the cyst were confirmed. A functional splenic parenchyma was documented 18 months after surgery. There was no infection during follow-up. Partial surgical splenectomy with resection of the entire cyst prevents recurrence and preserves splenic function.

Background: The finding of adnexal masses during pregnancy is an exceptional event. Its reported incidence is less than 5% and most cases resolve spontaneously as the pregnancy progresses., Objective: Describe a case series of patients with adnexal mass and pregnancy., Material and Methods: We retrospectively reviewed the medical records of patients who had diagnosis of pregnancy and adnexal over a period of five years., Results: The incidence was 0.22%. The mean age was 26 +/- 7.3 years, mean gestational age was 17 +/- 6.6 weeks. The diagnosis was established by ultrasound (USG) in 95% of cases, 48% had cystic characteristics, the mean diameter of the tumor was 99 +/- 42 mm. Cistectomy was performed during pregnancy or trans-cesarean section in 30% and 58% of patients respectably. The mean tumor size was 118 mm (range 2 a 40 mm), weight 1,370 g (range 10 a 5,800 g). The most frequent histological diagnosis were serous cyst (40%), mature teratoma (28%), mucinous (6%), malignancy (4%). There were not complications related to the surgical procedure., Conclusions: The USG constitute a safe method for the diagnosis, but the image method with the highest positive predictive value is the MRI. Tumor markers (CA-125, AFP, GCH-B,DHL, ACE), are not useful during pregnancy. If the tumor doesn't achieve surgical criteria the recommended follow up is clinical observation and USG. If surgery is decided, it should be performed between 16 a 23 weeks of pregnancy, and it's recommended to send the tumor to histological diagnosis, in case of malignancy the surgery will continue according to the tumor stage. The time and delivery route will be decided by the obstetrician.

Background: In the group of adult patients being diagnosed from different neurological complaints frequency of pineal cyst occurrence is estimated at 0,1-4,9%. Frequency of its occurrence in pediatric population is unknown, however it is regarded as a common lesion at this age, more frequent in girls and older children. This structural lesion is usually asymptomatic, without sequels and clinical complications. However, neoplastic lesions may also localize in pineal cyst., Aim of the Study: Characterization of pineal cysts found in children diagnosed from different neurological diseases., Material and Methods: 45 children at the age range from 14 months to 17 years were hospitalized in the Department of Pediatric Neurology Chair of Pediatric and Adolescent Neurology in the years 2006-2010. MR examination with contrast (with the use of Siemens device 1,5 T) revealed lesions in pineal gland defined as pineal cysts. Predominance of girls was observed in the group (33/45). Additionally older children between 10 and 15 years of age were dominating (27/ 45). In 24/45 children serum tumor markers AFP and betaHCG were determined. Examined group of patients with clinical symptoms which indicated necessity of MR examination performance was very heterogenic., Results: The diameter of pineal cyst was between 3-10 mm in 40/45 children, 13 mm in 2 children and 11 mm in 3 children (most often 4-5 mm in 18 children). Usually this lesion accompanied other congenital or acquired CNS disorders. In 1/3 of children included to the study, headaches were cause of brain MR examination. In 15 children brain MR examination was performed due to epileptic paroxysmal events. No positive values of tumor markers were found in any of examined children., Conclusions: 1. Pineal cysts are common structural lesions in children hospitalized from different neurological symptoms. 2. Pineal cysts were usually found in children diagnosed because of headaches and epilepsy, and these were the most often final diagnosis. 3. In the examined group children over the age of 10 years and female sex predominate. 4. It is suspected that in majority of hospitalized patients pineal cyst was an incidental finding, with no association with clinical symptoms.

Aim: Owing to grave socio-medical consequences, polycystic ovary syndrome (PCOS) is a highly alarming endocrinopathy. For searching better therapeutics for PCOS, we present a rat model for PCOS using antiprogestin RU486 and evaluate the efficacy of noscapine for its treatment., Material and Methods: Female Wistar rats weighing 200+/-10 g bwt, exhibiting regular estrous cycle were administered an oral dose of RU486 [20 mg/kg body weight (bwt)/day] in olive oil for 13 consecutive days and compared with controls receiving 0.1 ml olive oil/100 g bwt/day. PCOS induced rats were administered varying dosing regimens of noscapine and were further compared with flutamide, the conventional drug for PCOS. Consecutively, vaginal smears and ovulation was noted and rats from all the groups were sacrificed and serum hormone levels for LH, FSH, PRL, estradiol, and testosterone were measured by enzyme-linked immunosorbent assay. Uteri and ovaries were excised free of adjacent tissue, weighed and further recruited for ascertaining ovary histologic parameters., Results: Our data go in accordance with previous studies where RU486 administered rats mimicked classical PCOS parameters seen in women. PCOS induced rats with ovulation blockade, persistent estrus and polycystic ovary resumed estrous cycle in 3-4 days post noscapine administration (120 mg/kg bwt/day). Folliculogenesis was followed by ovulation with reduced cystic manifestation and restored ovary and uterus weight. Biochemically, serum LH, PRL, estradiol, and testosterone concentration showed reduction while FSH and progesterone concentration increased significantly when compared with the conventional drug flutamide., Discussion: The amelioration of PCOS by noscapine is a novel observation that makes it a potential candidate for being a better therapeutic modality.

Background: The aim of this study was to compare results of the determination of carcinoembryonic antigen, carbohydrate antigens, alpha-fetoprotein, and human chorionic gonadotropin before and after surgical and pharmacologic treatment in patients with cystic echinococcosis (CE)., Methods: Serum samples were obtained from 40 CE patients (all with cysts in the liver) and from 10 sex- and age-matched healthy donors (control group). Serum samples were drawn (1) before (presurgical group) and after (postsurgical group, including a 3-month cycle of albendazole) surgical and pharmacologic treatment. Serum tumor markers were measured, and indirect hemagglutination assay was performed., Results: In 90% of confirmed cases of CE, indirect hemagglutination assay was positive. Mean (SD) serum CA19-9 concentrations for all patients in the presurgical and postsurgical groups were 45.1 +/- 30 kU/L and 17.02 +/- 11 kU/L, respectively. CA19-9 concentrations were significantly greater in CE patients in the presurgical compared with the control group. Also, increased CA19-9 concentrations decreased significantly in the postsurgical compared with the presurgical group., Conclusions: A significant decrease in serum CA19-9 concentrations after surgical and pharmacologic therapy was demonstrated in the clinical follow-up of patients with CE (patients were tested 3 months after surgery). If our findings are confirmed and more-sensitive methods are developed for measuring serum CA19-9 concentrations, new and interesting perspectives will be gained for the monitoring and treatment of patients with CE.

Objectives: The study was undertaken to evaluate selected blood coagulation factors in patients undergoing surgery, due to endometrial cysts and other ovarian benign cysts., Materials and Methods: Women involved in our study had not received any previous treatment for endometriosis and they had no history of any prior haemostatic disorders. Blood samples were collected before surgery and investigated for plasminogen, alpha2-antyplasmin, PAI-1 and tPA activity. As a control group, we have analyzed patients with benign gynecological diseases treated in our Department., Results: We have noticed higher mean concentration of plasminogen and alpha2-antyplasmin and lower mean concentration of PAI-1 and tPA activity in our patients in comparison with control group. Obtained results did not show any statistical significance., Conclusions: Our analysis of haemostatic factors in blood samples did not show coagulation disorders in patients with endometriosis. Maybe there are only local coagulation disorders in endometrial tissue and its surrounding. In our opinion this problem requires further research and taking into consideration other factors.