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Labrune syndrome is a rare white matter disease characterized by angiomatous leukoencephalopathy, diffuse intracranial calcifications and supratentorial and infratentorial parenchymal cysts. The clinical worsening is often related to cyst expansion, and surgery may be advocated for symptomatic management in about one third of cases. However, no consensus exists on the surgical timing, the most effective procedure, and the long-term results. Electronic databases PubMed/MEDLINE and Google Scholar were searched for studies published up to April 2022 using the search string (Labrune syndrome OR leukoencephalopathy with calcifications and cysts OR brain calcifications OR brain cysts) AND (therapy OR surgery). We found 28 studies in the literature, and we added a new case from our institution, comprising 37 patients. All the patients in this series underwent surgical intervention. We reviewed all the pertinent literature to discuss clinical-radiological features and etiopathogenesis, specifically addressing the surgical options, clinical results, and prognosis. Leukoencephalopathy with cerebral calcifications and cysts is a rare neurodegenerative disorder for which effective medical treatment is lacking. Surgery remains the only therapeutic option to control the disease to reduce the mass effect of growing cystic lesions. Almost half of the patients who underwent surgery required further approaches, with great concern for the associated disabilities. Several procedures have been described, with no evidence regarding which procedure is the most effective. Individual-based surgical planning must be advocated, tailoring the approach to limit side effects. Mini-invasive neuroendoscopic approaches may be considered to achieve satisfactory results. [ABSTRACT FROM AUTHOR]

To retrospectively determine the incidence of intraosseous mandibular lesions among patients who attended the University dental clinic in Tanzania A retrospective study of histological results of the intraosseous mandibular lesions diagnosed between 2016 and 2021. The information gathered included the age and sex of the patient, and histological diagnosis. Data analysis was done using Statistical Package for the Social Sciences version 26 computer program. Out of 1824 reports of patients with oral and maxillofacial lesions retrieved, 475 (26 %) involved the intraosseous part of the mandible. Benign tumors comprised 68.2 % and malignant tumors 14.1 %. Ameloblastoma accounted for 54.3 % of the benign tumors, osteosarcoma comprised 37.3 % of the malignant tumors, and dentigerous cysts made up 70.4 % of mandibular cysts. Intraosseous mandibular lesions are common among patients who were biopsied at the university dental clinic. Although there is no sex predilection, young adults are more affected than other age groups. The majority of the lesions are benign and malignant tumors. [ABSTRACT FROM AUTHOR]

Non-endodontic lesions (NEL) closely resemble lesions of endodontic origin. Its etiology can be odontogenic, non-odontogenic, neoplastic, or anatomic variations that can resemble inflammatory periapical lesions in the periapical area. Inflammatory periapical lesions are caused by pulpal pathoses and require endodontic treatment. Since numerous NEL may resemble inflammatory periapical lesions, they can lead to misdiagnosis and inappropriate management. Thus, a detailed review of the patients' medical and dental histories with clinical examination, including radiographic findings, is essential for the proper assessment of periapical lesions. Numerous cases of misdiagnoses of NEL have been reported in literature. Thus, this review aimed to strengthen the awareness of clinicians on periapical radiolucency, which may resemble inflammatory periapical lesions. [ABSTRACT FROM AUTHOR]

To investigate the impact of virtual monoenergetic images (VMI) from photon-counting detector CT (PCD-CT) on the enhancement and classification of renal cysts. Adults with renal cysts (≥7 mm) who received a triphasic examination on a clinical PCD-CT (120 kVp; IQ level 68) between July 2021 and March 2022 were retrospectively identified. Only non-enhancing cysts (enhancement<10 HU between unenhanced and venous phase at 70 keV) were included. VMI from 40 to 190 keV with increments of 10 keV were reconstructed from the venous phase. Enhancement was measured to classify each lesion as non-enhancing (<10 HU), equivocally enhancing (10–19 HU), and definitely enhancing (≥20 HU). Classification changes as a function of VMI were assessed. Pearson correlation coefficient, the Kruskal-Wallis and the Chi-square test were used. A total of 86 patients (mean age, 74 ± 9 years; 74 male) with 160 non-enhancing renal cysts (17.6 ± 10 mm) were included. CT attenuation of the cysts increased from higher to lower VMI levels with a mean attenuation of 4 ± 11 HU at 190 keV to 36 ± 17 HU at 40 keV. Mean attenuation of the renal parenchyma was 43 ± 4 HU at 190 keV and 414 ± 71 HU at 40 keV. No cyst exhibited enhancement from 70 keV to 190 keV. At 40, 50, and 60 keV, 35% (56/160), 29% (47/160) and 9% (15/160) of cysts showed equivocal and 46% (74/160), 10% (16/160), and 0% (0/160) definite enhancement, respectively. There was no significant influence of size (P =.13), cyst location (P =.9) and BMI (P =.19) on enhancement classification. VMI has a relevant impact on enhancement and classification of renal cysts with misclassification in a large number of cases at energy levels below 70 keV. [ABSTRACT FROM AUTHOR]

The Sistrunk procedure has long been the method of choice for treating patients with thyroglossal duct remnants (TDRs). However, the extent of the surgical resection in the suprahyoid segment of the TDR remains controversial, as this anatomical site is the origin of both disease recurrence and surgical morbidity. The aim of this two-centre retrospective cohort study was to investigate the outcomes of a modified muscle-sparing Sistrunk procedure in primary TDRs. The primary predictor was the surgical approach, and the outcome variable was the recurrence rate. A total of 110 consecutive patients (62 (56.4%) male, 48 (43.6%) female) who underwent a modified muscle-sparing Sistrunk procedure were included in the study. Their mean age at presentation was 26.9 ± 18.9 years. A modified muscle-sparing Sistrunk procedure using cold instruments, electrocautery, and a harmonic scalpel was performed in all patients. Recurrence was recorded in six (5.5%) patients. The median hospital stay was 2 days (range 1–7 days), and the median follow-up duration was 7 years (range 2–13 years). There was no significant difference in recurrence rate between the conventional and modified muscle-sparing Sistrunk procedure in primary TDRs. The study findings showed that the modified muscle-sparing Sistrunk procedure had low recurrence and complication rates in the context of primary TDR. [ABSTRACT FROM AUTHOR]

• Wild type cells show an upregulation of lysine methyltransferase-2 during encystation. • Downregulation of lysine methyltransferases-2 prevents encystation and cyst formation. • Interplay of deacetylases and methyltransferases highlights encystation's complexity. Histone post-translational modifications are extensively studied for their role in regulating gene transcription and cellular environmental adaptation. Research into these modifications has recently begun in the protozoan parasite Giardia lamblia , focusing on histone-modifying enzymes and specific post-translational changes. In the transformation from the trophozoite to the cyst form in the life cycle of this parasite, significant morphological and genetic alterations occur, culminating in the synthesis of cyst wall proteins responsible for forming the protective cyst wall. It has been previously demonstrated that histone deacetylation is required during encystation and that the enzyme lysine methyltransferase 1 is involved in the upregulation of encystation. Our study aims to extend the analysis to lysine methyltransferase 2 (GlKMT2) function. For this, two constructs were generated: one that downregulate the expression of GLKMT2 via antisense (glkmt2-as transgenic cells) and the other overexpressing GlKMT2 (glkmt2-ha transgenic cells). We found that the glktm2-as transgenic cells showed an arrest in progress at the late encystation stage. Consequently, the number of cysts produced was lower than that of the control cells. On the other hand, we found that the overexpression of GlKMT2 acts as a negative mutant of the enzyme. In this way, these glktm2-ha transgenic cells showed the same behavior during growth and encystation as glkmt2-as transgenic cells. This interplay between different enzymes acting during encystation reveals the complex process behind the differentiation of the parasite. Understanding how these enzymes play their role during the encystation of the parasite would allow the design of inhibitors to control the parasite. [Display omitted] [ABSTRACT FROM AUTHOR]

This study's objective is to determine if the combined use of bone substitutes and collagen membranes is related to worse postoperative outcomes after intra-osseous oral cystectomies. Additional risk factors mentioned in the existing literature were retrospectively analysed by reviewing the medical records and imaging tests of adult patients. Age, gender, smoking habit, cyst location, size, shape, histopathology, perilesional osteosclerosis and associated intracystic third molars were registered. The type of biomaterials applied and the number of bony walls remaining after surgery were also identified. All factors were analysed in relation to wound dehiscence and postoperative infection. Simple and multiple logistic regression analyses were performed to exclude possible confounding effects between the risk factors identified. In a sample of 211 cystectomies, guided bone regeneration was not significantly associated with infection, only the presence of impacted third molars was (p = 0.0219). This finding remained true, even in cysts larger than 25 mm. However, the risk of wound dehiscence was 4.9 times higher when biomaterials were applied (CI 95%, 1.6818–12.8274, p = 0.0014). In accordance with recent studies, these findings suggest that whenever guided bone regeneration might prove useful after cyst enucleation, avoiding chemically cross-linked membranes should be considered. [ABSTRACT FROM AUTHOR]

• Intracranial ependymal cysts are rare benign lesions. • May have different clinical presentations depending on its location. • Definitive diagnosis depends upon histopathological and immunohistochemical analysis. • Surgical management is currently the main therapy for symptomatic patients. • Surgery is needed in patients with a focal neurological deficit. Intracranial ependymal cysts (IECs) are rare, histologically benign neuroepithelial cysts that mostly occur in the cerebral parenchyma. The majority of these cysts are clinically silent and discovered incidentally, but when symptomatic they may compress surrounding structures, thus surgical intervention is needed. The current data in the literature about ECs is very scarce, and many are misdiagnosed, once they share many radiological characteristics with a variety of intracranial benign cysts. Also their terminology is confusing, and its definitive diagnosis can only be made through a thorough histopathological study, hence a detailed description about these uncommon lesions is necessary. The correct identification of the lesion lead to our better understanding of the condition and further improvement of the patient's prognosis. A descriptive case is presented; moreover, a detailed PubMed search according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guideline was performed. The data found was analyzed by various criteria in order to correctly describe the characteristics of this lesion. The literature review gathered 9 descriptions of patients with IECs with a diverse range anatomopathological and clinical manifestations. All of the included studies found were case reports. Moreover, the authors suggest an updated classification of the lesion, involving their immunohistochemical characteristics. The information obtained from this study highlights IECs rarity and their inaccurately classification. We propose that the definitive diagnosis of IECs shall be made upon histopathological confirmation of an ependyma-lined cyst along with a positive glial fibrillary acidic protein (GFAP). [ABSTRACT FROM AUTHOR]

To provide data on the measurable vocal fold vibratory differences in children with and without vocal fold lesions using high-speed videoendoscopy. Prospective study, 24 participants (8 healthy; 16 with lesions) between the ages of 5 and 10. Rigid high-speed videoendoscopy at the rate of 8,000 frames per second was used to examine participants. Four objective vocal fold phase linearity measures were obtained to establish anterior-posterior contact and separation vibratory patterns. All objective measures showed a difference between nonlesion and bilateral vocal fold lesion groups. Contact-separation patterns in all nonlesion girls and young pre-pubertal boys exhibited an anterior-to-posterior contact and posterior-to-anterior separation; while older boys differed. The objective measures of open quotient, left-right relative phase asymmetry and speed index, showed linear anterior-posterior patterns within the nonlesion group; while the bilateral vocal fold lesion group displayed nonlinear patterns. Patterns in the posterior region of the vocal fold were similar in both groups; while patterns in the anterior region differed. This study suggests lesions have an effect on the anterior aspect of vocal fold vibratory patterns specifically anterior to the lesions. Age-related differences for males are also evidenced, prompting further investigation of laryngeal development in males and females from childhood to adulthood. This study could serve as a basis for the development of objective clinical measurements of vocal fold vibration in presence of lesions. Further findings could help redefine the theoretical framework of pediatric voice. [ABSTRACT FROM AUTHOR]

The earliest reported case of the occurrence of a dentigerous cyst is described; the cyst surrounded an unerupted permanent tooth bud in a 6-month-old infant. Most commonly these lesions present between the second and third decades of life. They rarely occur before 10 years of age and have not been documented prior to 1 year of age. In the case reported here, the treatment instituted was extraction of the adjacent deciduous tooth and enucleation of the cyst along with the permanent molar tooth bud. Clinicians should be aware of the potential for this lesion to occur across a wide range of ages and the importance of prompt diagnosis and treatment to prevent complications and reduce morbidity. [ABSTRACT FROM AUTHOR]

To evaluate the relationships between calibration dose rate, calculated biologically effective dose (BED), and clinical factors and tumor control after stereotactic radiosurgery (SRS) for acoustic neuroma. We performed a retrospective study of all patients with acoustic neuromas treated with frame-based cobalt-60 SRS at a single institution between 2005 and 2019. The calibration dose rate and cobalt-60 half-life were used to calculate the nominal dose rate during treatment. An SRS-specific monoexponential model accounting for treatment time per lesion was used to estimate BED. The study included 607 patients with 612 acoustic neuromas. Median follow-up was 5.0 years. There was no association between dose rate or BED with local failure (LF), radiologic edema, or symptomatic edema. Cystic tumors (adjusted hazard ratio 0.26, P = 0.028) were associated with lower LF, while use of SRS as salvage treatment for growing tumors (adjusted hazard ratio 4.9, P < 0.0001) was associated with higher LF. LF occurred more frequently in larger-diameter tumors, while radiologic or symptomatic edema occurred more frequently in larger-volume tumors. Radiosurgery dose rate and BED were not associated with tumor control or radiologic or symptomatic edema. Salvage SRS and larger tumors were associated with a higher LF rate, while cystic tumors were associated with a lower LF rate. Patients with larger tumors should be counseled appropriately about potential side effects and when to seek follow-up care. [ABSTRACT FROM AUTHOR]

To evaluate the morphologic characteristics and clinical significance of epidural gas based on computed tomography (CT) and magnetic resonance imaging (MRI) findings and to determine their relationship with radiculopathy. Between March 2009 and November 2018, 110 epidural gas lesions were identified in 103 patients who underwent both CT and MRI for suspected herniated disc in the authors' institution. Patterns of epidural gas were classified as air pseudocyst, air cyst, air-contained disc herniation, and honeycomb-like air cyst. These gas patterns were compared, and possible correlations between these gas patterns and radiculopathy were evaluated. Overall agreement between CT and MRI findings for evaluation of all lesions and for differentiation of epidural gases was good (kappa [κ] = 0.775). Air pseudocysts demonstrated a moderate correlation (κ = 0.496) and air cysts showed a good correlation (κ = 0.661) with radiculopathy on MRI, whereas air-contained disc herniation and honeycomb-like cysts demonstrated a strong correlation (κ = 0.810 and 0.927, respectively) with radiculopathy on MRI. This study's results help delineate new classifications of epidural gases. In addition, lumbar epidural gas with disc material (e.g., air-contained disc herniation and honeycomb-like cysts) on MRI was associated with radiculopathy. [ABSTRACT FROM AUTHOR]

The recent detection of a novel amoebozoan parasite (Entamoeba sp. CT1) killing invasive cane toads (Rhinella marina) in tropical Australia raises concerns of potential spill-over into native anuran populations. Considering the vulnerability of anuran communities globally, Entamoeba sp. CT1 may pose a serious threat to anuran biodiversity. Through PCR-based detection and molecular identification, we investigated the prevalence of Entamoeba spp. in the faeces and colon tissue of cane toads (Rhinella marina) and eleven native Australian frog species from a single locality in the Northern Territory. No Entamoeba DNA was detected in samples of native frog faeces (N = 57) or colons (N = 17). Entamoeba DNA was detected in 24% of 45 cane toads (95%CI 14.08–38.82). Both E. ranarum and Entamoeba sp. CT1 were present in cane toads. The failure of faecal samples to indicate Entamoeba spp. in infected cane toads may be due to cysts in faeces being shed intermittently, degraded before analysis, or impervious to lysis prior to DNA isolation. Our results suggest that native frogs do not carry the pathogen in an area where 20–30% of cane toads are infected with Entamoeba sp. CT1. We demonstrate the importance of recognising PCR inhibition prior to molecular diagnostics, and the apparent inadequacy of faecal samples for the detection of Entamoeba spp. in anurans. Image 1 • Entamoeba spp. were detected in cane toads, but not in faeces or colons of native frogs. • PCR inhibition of anuran faecal samples. • Additional purification, the presence of inhibition was reduced. [ABSTRACT FROM AUTHOR]

• Large hepatic cysts may be at risk of infection following pancreatoduodenectomy. • Percutaneous transhepatic drainage, rather than antibiotic administration, is essential treatment. • Minocycline hydrochloride injection into the infected cyst is also effective. Asymptomatic hepatic cysts, often observed on computed tomography, have never been reported as a risk factor for infection following pancreatoduodenectomy. Symptomatic liver cysts are treated surgically or non-surgically. We encountered a case of infected hepatic cyst following pancreatoduodenectomy. An 88-year-old woman underwent pancreatoduodenectomy for adenocarcinoma in the duodenum papilla of Vater. She was discharged (postoperative day 23), following an uneventful course. However, 7 days later, she was re-admitted because of a high fever; postoperative cholangitis was suspected. A peripheral-venous blood bacterial culture was negative. Her C-reactive protein level and white blood cell count were 0.57 mg/dl and 5290/μl, respectively. Antibiotics were administered, but her high fever only temporarily decreased. Contrast-enhanced computed tomography showed ring enhancement surrounding the largest of several hepatic cysts. Percutaneous transhepatic drainage was performed; thereafter, minocycline hydrochloride was repeatedly injected into the infected cyst through the drain to prevent infection recurrence. The drain was removed 49 days after drainage. On contrast-enhanced computed tomography at 6 months postoperatively, the infected hepatic cyst had reduced in size from 6.0 to 1.7 cm and no findings of bile duct stenosis were observed. No further infectious events have been observed. Relatively large hepatic cysts may be at risk of infection following pancreatoduodenectomy and, in particular, biliary reconstruction. Transhepatic drainage, rather than antibiotic administration, is essential, and minocycline hydrochloride injection into infected cyst that have no evidence of communication with the biliary tree is effective in preventing infection recurrence, without the complication of bile duct stenosis. [ABSTRACT FROM AUTHOR]

Vestibular schwannomas (VSs) can either be solid or contain variable amounts of cystic components. It has been established that gamma knife radiosurgery (GKRS) is an effective treatment modality for solid VSs. However, in the case of cystic VS, given the somewhat unpredictable growth of the cystic portion of these tumors, microsurgery has often been preferred to GKRS in the management of cystic VS. However, to date, a relative paucity of data evaluating the outcomes after GKRS for cystic VS has been available. In the present study, we reviewed our experience treating cystic VSs with GKRS and evaluated the results. The data from patients with a diagnosis of cystic VS who had undergone GKRS from 1999 to 2018 were analyzed. Only those patients who had undergone GKRS as the initial treatment of cystic VSs were included in the present study. Patients who had undergone previous surgical treatment were excluded. The cystic VSs were divided into intratumoral and peritumoral cysts. A total of 24 patients were enrolled in the present study. The mean age was 60.8 ± 2.4 years (range, 38–82 years). The mean follow-up period was 55.8 ± 8.1 months (range, 8–145 months). Of the 24 patients, 11 were men. The control rate after GKRS for cystic VSs was 75%. The 5-year progression-free rate was 70%. Six patients had presented with an increasing lesion size after GKS. Of these 6 patients, 2 underwent surgical treatment and 1 underwent repeat GKRS. No statistically significant differences were found between the control rate and cystic type (P = 0.093). The results from the present study have demonstrated that GKRS is an effective treatment modality for cystic VSs. [ABSTRACT FROM AUTHOR]

• Indocyanine green helps in laparoscopic surgery for type II cystic biliary atresia. • Fluorescence imaging can replace cholangiography while avoiding radiation. • Indocyanine green can be used for intraoperative navigation in Kasai surgery. In recent years, it has been reported that indocyanine green can be used for intraoperative navigation in Kasai surgery. However, there are no reports of its application in surgery for rare type II cystic biliary atresia. We report a girl presented with jaundice and light-colored stools. Laboratory tests showed impaired liver function with elevated serum bilirubin and bile acid levels. The abdominal ultrasound and MRCP suggested a common hepatic duct cyst. A diagnosis of choledochal cyst was suspected and biliary atresia could not be excluded. Conservative treatment was unsatisfactory. Laparoscopic exploration with indocyanine green fluorescence was performed on the 38th day of her life, and intraoperative diagnosis of type II CBA was made because the common hepatic duct cyst and its downstream anatomical structures did not show fluorescence. The postoperative bilirubin and bile acid levels decreased significantly and she was discharged two weeks after surgery. This result suggests that indocyanine green can be safely used in laparoscopic surgery for type II CBA, which not only helps in the differential diagnosis of CBA and choledochal cyst, but also confirms bile flow in real time. [ABSTRACT FROM AUTHOR]

Ovarian lesions are the most common abdominal masses in the pediatric population yet have varied presentation. They are often missed or misdiagnosed causing delay in management and complications. Knowledge gained in managing ovarian lesions in one age group cannot be extrapolated to another group. This paper reviews the common and uncommon clinical presentations of ovarian lesions according to age aiming at timely diagnosis and appropriate management. The review was done according to the PRISMA 2020 guidelines. PubMed and Scopus were systematically searched using the key words clinical characteristics or clinical features of ovarian masses, cysts, tumors and torsion in neonates, premenarchal and adolescent girls from 2008 to 2022. The inclusion criteria were full text articles in English, female and human species. Articles with incomplete data, that were non-specific for age, with no sample size and without open access were excluded. The study is approved. Seventy-eight studies were included. 57 from PubMed, 21 from Scopus. Neonates numbered 24. All presented with ovarian cysts which were discovered antenatally on routine ultrasound. Postnatally, none had symptoms, except one who presented with lethargy and emesis. Abdominal fullness and mass were common signs. Uncommon findings were intestinal obstruction, hydronephrosis, breast budding and pubic hair. 33% of the neonates developed ovarian torsion. The premenarchal group had 112 participants. Abdominal pain, nausea, vomiting, anorexia and fever were common symptoms. Breast budding, pubic hair development, vaginal bleeding and growth spurts were also commonly noted. Less commonly were symptoms of urinary retention, constipation and intestinal obstruction. The adolescent group had 836 participants – the largest of the three groups. The ovarian lesions in this group comprised of tumors with symptoms of abnormal uterine bleeding, abdominal pain, nausea and fever. Less commonly were symptoms of precocious puberty and hydronephrosis. Neonates uncommonly present with symptoms of ovarian lesions. Therefore, a high index of suspicion is needed when one encounters a neonate with an abdominal mass. Those discovered antenatally should have routine ultrasound follow up postnatally to reduce the incidence of ovarian torsion. Ovarian lesions in the premenarchal group were largely misdiagnosed causing delayed treatment and torsion, making ovarian sparing surgery difficult. Those presenting with precocious puberty should be evaluated for functional ovarian masses. Adolescent girls with menstrual irregularities and elevated tumour markers should also be assessed for ovarian masses. Timely diagnosis of ovarian masses in children leads to early and appropriate management with fewer complications and fertility sparing surgery. ▪ [ABSTRACT FROM AUTHOR]

• Leukoencephalopathy, cerebral calcifications, and cysts (LCC) is a rare disease. • TLCC shows distinct triple imaging findings in the supra- and infratentorial brain. • LCC involves microangiopathy, serum collection, and dystrophic calcification. • The clinical features of LCC depend on the mass effect and hemorrhages. • Late recurrence of cysts and leukoencephalopathy warrants long-term follow-up. Leukoencephalopathy, cerebral calcifications, and cysts (LCC) is an extremely rare neurological disease, also known as Labrune syndrome. The disease more commonly affects children and young adults and the characteristic triple imaging findings are leukoencephalopathy, calcifications and multiple cysts, presenting with a variety of supra- and infratentorial symptoms but lacking for extra-neurological manifestations. Coats plus syndrome and cerebroretinal microangiopathy with calcifications and cysts (CRMCC) share similar neurological findings with LCC, but additionally involves other extra-neurological organs. Tumoral excision is usually required due to mass effect to the eloquent brain of multiple growing cysts or hemorrhages, but the outcome of surgery varies. Here we demonstrate an 8-year neuroimaging study of a rare adult-onset case of LCC with gradual headache, hemiparesis, hand tremors, unstable gait, and seizure attacks despite several times of tumoral excision. Neuroimaging revealed multiple microbleeds and microcalcification in the leukoencephalopathic areas, with increasing calcifications, recurrent previously excised cysts and new cyst formation in the longitudinal neuroimaging follow-ups within the eight years. We believe that LCC involves microangiopathy, which causes blood-brain barrier disruption, myelin serum collection and subsequent growing cysts and dystrophic calcification formation. We provide histopathological correlation in the illustration. Due to the underlying pathomechanism and long-term recurrence nature, patients with a combination of cysts and calcifications on CT scan should be follow up carefully and postoperative recurrence after years may occur. [ABSTRACT FROM AUTHOR]

Cyst abundance and identity are essential for understanding and predicting blooms, and for assessing the dispersal of toxic target dinoflagellate species by natural or human mediated ways, as with ballast waters. The aim of this study was to apply rapid, specific and sensitive qPCR assays to enumerate toxic dinoflagellate cysts in sediment samples collected from Adriatic harbours. The molecular standard curves of various target species allowed obtaining the rDNA copy number per cyst. The analytical sensitivity for specific standard curves was determined to be 2 or 10 rDNA copies per reaction. The abundance varied in the range of 1–747 dinoflagellate cysts g−1 dry weight. The assays showed greater sensitivity as compared to counts by light microscopy. This qPCR method revealed a powerful tool for the quantification of cysts from toxic dinoflagellate resting stages in sediment samples from Adriatic ports. Unlabelled Image • Cysts of toxic dinoflagellate species can be dispersed by ballast waters in the Adriatic ports. • We applied qPCR assays to monitor and quantify cysts from toxic species in sediment from ports. • The quantification was done by species-specific standard curves. • The qPCR assays can be applied in monitoring activities to assess environmental port quality. [ABSTRACT FROM AUTHOR]

Cystic lung diseases are a group of rare disorders that can solely involve the lungs or might be part of a multi-organ process. Multidisciplinary assessment, including lung histology, is key to reaching the final diagnosis, as some might be hereditary and linked to a risk of malignancy. Tissue for microscopic examination of the lungs is mainly accessed during surgical treatment for pneumothorax, and can suggest an underlying cystic disease. This multidisciplinary approach is reflected in this review article, with summaries of the clinico-radiological presentation of cystic lung disease and detailed histopathological descriptions of these uncommon conditions, namely lymphangioleiomyomatosis, pulmonary Langerhans cell histiocytosis, Birt-Hogg-Dubé syndrome, lymphocytic interstitial pneumonia, pulmonary amyloid and light chain deposition disease and metastases of sarcomas. [ABSTRACT FROM AUTHOR]

• A mouse model for Giardia duodenalis cyst propagation was developed as an alternative to a gerbil model. • The mouse model had a continuous patency period in contrast to the cyclical pattern in gerbils. • The mouse derived cysts were genetically stable to gerbil derived cysts based on β- giardin sequence. This study developed and evaluated Giardia duodenalis cyst propagation using a dexamethasone immunosuppressed CF-1 mouse model as an alternative to a previously described Mongolian gerbil model. The CF-1 mouse model shed significantly more cysts per animal during a 16–18 h collection period compared to the gerbil (averages: 7.8 × 106 cysts/CF-1 mouse and 2.5 × 106 cysts/gerbil). In addition, the patency period for this model differed from both G. muris in mice and G. duodenalis in gerbils in that cysts were shed continuously for over 20 days. Results further showed that the β- giardin gene sequences from gerbil derived and mouse derived G. duodenalis were identical, after 34 serial passages through the CF-1 mouse model. Overall, the CF-1 mouse model produced higher concentrations of cysts per animal, and were genetically and phenotypically stable based on β- giardin gene sequences. [ABSTRACT FROM AUTHOR]

Abstract Autosomal dominant polycystic kidney disease (ADPKD) is caused by mutations in genes encoding the polycystin (PC) 1 and 2 proteins. The goal of this study was to determine the role of calcium in regulating cyst growth. Stromal interaction molecule 1 (STIM1) protein expression was 15-fold higher in PC1-null proximal tubule cells (PN) than in heterozygote (PH) controls and 2-fold higher in an inducible, PC1 knockout, mouse model of ADPKD compared to a non-cystic match control. IP3 receptor protein expression was also higher in the cystic mice. Knocking down STIM1 with siRNA reduced cyst growth and lowered cAMP levels in PN cells. Fura2 measurements of intracellular Ca2+ showed higher levels of intracellular Ca2+, SOCE and thaspigargin-stimulated ER Ca2+ release in PN vs. PH cells. There was a dramatic reduction in thapsigargin-stimulated release of ER Ca2+ following STIM1 silencing or application of 2-APB, consistent with altered ER Ca2+ movement; the protein expression of the Ca2+-dependent adenylyl cyclases (AC) AC3 and AC6 was up- and down-regulated, respectively. Like STIM1 knockdown, application of the calmodulin inhibitor W7 lowered cAMP levels, further indicating that STIM1 regulates AC3 via Ca2+ We conclude that the high levels of STIM1 in ADPKD cells play a role in supporting cyst growth and promoting high cAMP levels and an increased release of Ca2+ from the ER. Thus, our results provide novel therapeutic targets for treating ADPKD. Graphical abstract Unlabelled Image Highlights • Store operated calcium entry is elevated in ADPKD. • ER calcium release is elevated in PC1 null cells. • Silencing of high levels of STIM1 in ADPKD cells reduces cysts, cAMP and ER calcium release. • High levels of intracellular calcium activate adenylyl cyclase 3 fueling cyst growth. [ABSTRACT FROM AUTHOR]

Gamma Knife ventral anterior capsulotomy is an effective option to treat refractory obsessive-compulsive disorder. Although well tolerated, complications can develop years after radiosurgery. We describe a case in which abnormal complications induced by very high doses of radiation evolved. A 55-year-old man with refractory obsessive-compulsive disorder was treated with Gamma Knife ventral anterior capsulotomy using a dose of 180 Gy. His obsessive-compulsive symptoms improved, but his condition evolved with a manic episode, cognitive memory changes, visual hallucinations, confabulation, and frontal lobe symptoms. Magnetic resonance imaging showed brain edema in the left hemisphere and a 6-mm brain cyst in the right hemisphere at postoperative month 20. This case shows the consequences of affecting more fibers related to the anterior frontal region than intended with a radiosurgical procedure and illustrates the importance of careful clinical and imaging follow-up after Gamma Knife ventral anterior capsulotomy. [ABSTRACT FROM AUTHOR]

Information on fish reproductive strategy is essential to understand population dynamics. Samples of black seabream, Spondyliosoma cantharus , were collected from the western Portuguese continental coast to investigate the reproductive cycle, the timing of spawning, length at maturity and sex change, fecundity type and fecundity. This species is a protogynous hermaphrodite, showing a sharped biased sex ratio towards females for lengths smaller than 25 cm, and significantly biased towards males above this value, with no females occurring above 35 cm. Development of secondary growth follicles was asynchronous, and it was estimated that each female spawns 27 batches during the spawning season, which takes place from February to May. Half of the females’ population was mature at 18.41 cm and have changed sex at 25.62 cm. Sex change takes only a brief period of time, as transitional individuals were scarce and most showed oocytes regressing into cystic structures. The species presents a clear indeterminate fecundity type with massive atresia happening at the end of the spawning season. Mean values of 203 oocytes and 5431 oocytes by gram of eviscerated female were estimated for relative batch fecundity and relative annual fecundity, respectively. [ABSTRACT FROM AUTHOR]

Abstract Background Patients with cystic fibrosis (CF) and a CFTR gene mutation may present with a variety of pancreatic disorders. The presence of multiple macrocysts (>1 cm) replacing the entire pancreatic parenchyma is termed pancreatic cystosis. Lack of clear data makes clinical decision making challenging and controversial. The aim of this review is to perform a qualitative systematic analysis of the literature with intention to evaluate management plans. Methods Electronic databases MEDLINE, Embase, and Scopus were searched for relevant studies, and 19 studies describing patients with pancreatic cystosis were included and analyzed for clinical features and therapy offered. Results The data of 24 patients were collected from included studies. Eight cases (33%) had a documented CFTR gene mutation and 10 (42%) were symptomatic at presentation. Imaging modalities included ultrasound in 18 (75%), CT in 12 (50%), and MRI in 8 (33%) cases. An average size of the largest cyst was 5.4 cm. 6 (25%) patients were offered therapy that described surgical (3), endoscopic (1), or medical therapy (2). Surgeries offered included total pancreatectomy, partial pancreatic resection of uncertain extent, and complex cyst resection. Endoscopic treatment was cystogastrostomy. Novel medical treatment was utilized with Doxepin, Propantheline, and Clonidine, resulting in reduction in cyst size and overall clinical improvement. Conclusion Patients with pancreatic cystosis should not be denied treatment when necessary. This literature review is the most comprehensive thus far of cystic fibrosis and pancreatic cystosis, and it did not provide identification of a definitive treatment plan or demonstrate contraindication to specific therapies. [ABSTRACT FROM AUTHOR]

Azotobacter vinelandii is a γ-proteobacterium belonging to the Pseudomonadaceae family, that is capable of developing a differentiation process leading to the formation of cysts resistant to desiccation. The morphological, metabolic and biochemical changes occurring during encystment have been extensively studied. To elucidate the molecular basis of this process, the proteome of cells undergoing encystment was determined by quantitative proteomic analysis by LC-MS/MS. We identified 312 proteins differentially expressed after 48 h of encystment induction, with respect to vegetative cells. In agreement with the biochemical and physiological changes known to occur during this process, we found modifications in the abundance of proteins involved in nitrogen fixation, synthesis of the flagella, cell division, the glyoxilate shunt and in the synthesis of phenolic lipids known to replace the membrane phospholipids during cyst formation. In addition, our analysis also identified proteins for other cellular processes, probably important for desiccation resistance, such as the synthesis of trehalose. Proteins previously identified as critical for cyst development, also exhibited differences in abundance during encystment induction. This work represents the first study of the global changes in protein abundance during cysts development, and contributes to a better understanding of the molecular mechanism underlying this differentiation process. Biological significance Azotobacter vinelandii has been an important model for the study of the cyst developmental process in Gram-negative bacteria. In the current study, we have used a large-scale proteomic approach, using quantitative proteomic analysis by LC-MS/MS, to document changes in protein abundance in A. vinelandii cysts, relative to vegetative growing conditions. A total of 312 proteins were identified with differences in their abundance after 48 h of encystment induction. Remarkably, 62 proteins were found exclusively in the differentiated cell. As expected, some of the identified proteins were related to processes previously reported to occur during cyst development. However, the predicted functional categorization also revealed other biological processes likely important for the successful formation of the cyst. We believe that these novel findings not only help us to better understand the molecular basis of the developmental life cycle of A. vinelandii , but also the global molecular changes occurring during cyst formation in other Gram-negative bacteria. [ABSTRACT FROM AUTHOR]

Bisphenol A (BPA) has been well documented for its endocrine disrupting potential however, very little is known about endocrine disrupting abilities of bisphenol S (BPS). The present study aimed to compare the endocrine disrupting potentials of BPS with BPA, using female rats as an experimental animal model. On postnatal day 1 (PND 1) female pups born were randomly assigned to seven different treatments. Control group received subcutaneous injection of castor oil (50 μL) from PND 1 to PND 10. Three groups of female pups were injected subcutaneously with different concentrations (0.5, 5 and 50 mg/kg in 50 μL castor oil) of BPS, while remaining three groups were treated with 0.5, 5 and 50 mg/kg BPA. Highest doses treatments of both compounds resulted in delayed puberty onset and altered estrous cyclicity. Final body weight was significantly high in the highest dose treated groups of both BPS and BPA. Gonadosomatic index, absolute and relative weight of uteri was significantly reduced in BPS (5 and 50 mg/kg) and BPA (5 and 50 mg/kg) treated groups than control. Plasma concentrations of testosterone and estradiol were significantly increased, while plasma progesterone, Luteinizing hormone (LH) and Follicle stimulating hormone (FSH) concentrations were significantly reduced in highest doses treated groups. Dose dependent increase in the number of cystic follicles in the ovaries was evident along with an increase in the number of atratic follicles. The results suggest that neonatal exposure to higher concentrations of BPS can lead to BPA like structural and endocrine alterations in female rats. [ABSTRACT FROM AUTHOR]

Macular edema consists of intra- or subretinal fluid accumulation in the macular region. It occurs during the course of numerous retinal disorders and can cause severe impairment of central vision. Major causes of macular edema include diabetes, branch and central retinal vein occlusion, choroidal neovascularization, posterior uveitis, postoperative inflammation and central serous chorioretinopathy. The healthy retina is maintained in a relatively dehydrated, transparent state compatible with optimal light transmission by multiple active and passive systems. Fluid accumulation results from an imbalance between processes governing fluid entry and exit, and is driven by Starling equation when inner or outer blood-retinal barriers are disrupted. The multiple and intricate mechanisms involved in retinal hydro-ionic homeostasis, their molecular and cellular basis, and how their deregulation lead to retinal edema, are addressed in this review. Analyzing the distribution of junction proteins and water channels in the human macula, several hypotheses are raised to explain why edema forms specifically in the macular region. "Pure" clinical phenotypes of macular edema, that result presumably from a single causative mechanism, are detailed. Finally, diabetic macular edema is investigated, as a complex multifactorial pathogenic example. This comprehensive review on the current understanding of macular edema and its mechanisms opens perspectives to identify new preventive and therapeutic strategies for this sight-threatening condition. [ABSTRACT FROM AUTHOR]

Background The history of total ankle arthroplasty (TAA) has different evolution steps to improve the outcome. The third generation implants show an overall 8-year survival rate up to 93%. The main reported reason for early failure of TAA is aseptic loosening, cyst formation is also frequently reported. The aim of the present study is to use the finite element (FE) method to analyze the adaptive bone remodeling processes, including cyst formation after TAA. Methods Bone characteristics applied to the model corresponded to information obtained from computed tomography. Finite element models for the tibia and the talus were developed and implant components were virtually implanted. Results The calculated total bone loss is 2% in the tibia and 17% in the talus. Cysts and areas of increased bone density were detectable dependent on prosthesis design in the tibia and talus. Conclusion Our FE simulation provides a theoretical explanation for cyst formation and increasing bone density depending on implant design. However, cysts are not mono-causal, histo-chemical reactions should also be considered. Further clinical studies are necessary to evaluate the relevance of cyst formation and therapeutic strategies. [ABSTRACT FROM AUTHOR]

• Metabolomic profiling can identify biomarkers in patients with echinococcosis. • Alveolar- (AE) and cystic- echinococcosis (CE) biomarkers differ. • Inactive AE lesions alter amino acid metabolism. • Inactive CE lesions alter oxidative stress response metabolism. Hepatic alveolar echinococcosis (AE) and cystic echinococcosis (CE) are severe helminthic zoonoses and leading causes of parasitic liver damage. They pose a high mortality risk due to invisible clinical signs, especially at the early inactive stage. However, the specific metabolic profiles induced by inactive AE and CE lesions remain largely unclear. Therefore, we used gas chromatography-mass spectrometry-based metabolomic profiling to identify the global metabolic variations in AE and CE patient sera to differentiate between the two diseases and reveal the mechanisms underlying their pathogenesis. In addition, specific serum biomarkers of inactive hepatic AE and CE were screened using receiver operating curves, which can contribute to the clinical diagnosis of both diseases, especially in the earlier phase. These differential metabolites are involved in glycine, serine, tyrosine, and phenylalanine metabolism. Further analysis of key metabolic pathways showed that inactive AE lesions strongly alter amino acid metabolism in the host. CE lesions have an altered metabolism of oxidative stress response. These changes suggest these metabolite-associated pathways can serve as biomarkers to distinguish individuals with inactive AE and CE from healthy populations. This study also investigated the differences in serum metabolic profiles in patients with CE and AE. The biomarkers identified belonged to different metabolic pathways, including lipid, carnitine, androgen, and bile acid metabolism. Taken together, by investigating the different phenotypes of CE and AE with metabolomic profiling, serum biomarkers facilitating early diagnosis were identified. [ABSTRACT FROM AUTHOR]

Background Coats plus syndrome is a recently described, very rare multisystem disorder. The clinical phenotype is wide and variable, which making the diagnosis more difficulty. The genetic study of Coats plus syndrome has been reported recently. The biallelic heterozygous mutations in CTC1 gene, encoding conserved telomere maintenance component 1, were identified in families with Coats plus from different ancestry (European, American, and African). To data, there has not been a report about genetically confirmed Coats plus syndrome from China. Results We firstly identify a novel biallelic heterozygous missense variants (c.775G > A p.V259M and c.2066A > G p.Y689C) of CTC1 gene in a Chinese family with Coats plus. The c.2066A > G mutation (p.Y689C) in CTC1 is a novel variant. Such variant was not found in any of the 85 healthy individuals in the same community. Conclusion This is the first report of a genetically confirmed case of Coats plus from China. Targeted sequencing of CTC1 gene is useful for genetic diagnosis in Coats plus and differential diagnosis for other patients with similar disease manifestations. [ABSTRACT FROM AUTHOR]

Adult-onset leukoencephalopathies with increased cerebral volume can present a potentially challenging diagnosis for the pathologist. We present the case of a patient with a rare adult-onset disease called Leukoencephalopathy with cerebral Calcifications and Cysts (LCC). A 55-year-old woman with a history of morning headaches, mild memory loss, diabetes, and hypertension presented to the emergency department with acute onset altered mental status. CT scan revealed multiple small hypodense lesions in the white matter with calcifications in the bilateral cerebral hemispheres, basal ganglia, pons, and cerebellar hemispheres. MRI showed multiple complex/hemorrhagic cystic lesions with partial enhancement in addition to calcifications bilaterally in the frontotemporal white matter, pons, and cerebellar hemispheres, and diffuse white matter signal abnormality. The differential diagnosis included chronic infection, chronic thromboembolic disease, and neoplasm. The biopsy revealed extensive geode-like mineralization as well as smaller calcifications (calcospherites) with associated sclerosis, Rosenthal fibers, angiomatous proliferation of blood vessels with thrombosis and microbleeds. We discuss the differential diagnosis, radiologic and detailed histologic features of LCC. [ABSTRACT FROM AUTHOR]

Summary Cubital tunnel syndrome is the second most common nerve compression syndrome in peripheral nerve compression disease. Although potential ulnar nerve entrapment can occur at multiple points along its course, such as the arcade of struthers, the medial intermuscular septum, the medial epicondyle, the cubital tunnel, and the deep flexor pronator aponeurosis, the most common site of entrapment is the cubital tunnel. However, cubital tunnel syndrome could also be caused by the occupying masses along the course of ulnar nerve, such as intraneural or extraneural ganglia. The cubital tunnel syndrome caused by intraneural or extraneural ganglion cysts has been rarely reported. In our hospital, there were 184 patients with cubital tunnel syndrome who underwent surgical treatment from January 2010 to January 2014. Of these patients, 16 had extraneural cysts and 3 had intraneural ganglion cysts. The incidence rate of cysts in the cubital tunnel was 10.33%. Electromyography was used as routine examination. Ultrasound was used only in some patients in whom elbow mass was suspected. In the surgery of the cubital tunnel syndrome combined with cyst, if any other cysts were found, we should be remove completely the cyts and decompress the ulnar nerve thoroughly with the ulnar nerve being anterior transposition. These cysts were confirmed by histopathological examination. Finally, we compared the clinical features of patients who had a medial elbow ganglion with those of patients who had only cubital tunnel syndrome. B ultrasound can significantly improve the diagnosis. All patients were followed up for 4 months to 2 years, and the curative effect was good. [ABSTRACT FROM AUTHOR]

The keratocystic odontogenic tumour (KCOT) is one of the most aggressive odontogenic cysts and has a high recurrence rate. The treatment of these tumours is the subject of debate. A KCOT in the posterior maxilla with sinus involvement is rare. Few reports have been published in the literature. The purpose of this study was to evaluate the recurrence rate after surgical removal of maxillary KCOTs via a Le Fort I osteotomy. A search was performed to identify patients with a follow-up time of at least 5 years. Nine patients were included in the study. The following clinical variables were analyzed: age at surgery, sex, symptoms, site and size of the tumour, surgical approach, and recurrence rate. The surgical approaches were curettage ( n = 6) and enucleation ( n = 3). Recurrence was seen in three patients (33%); all had multilocular tumours. No recurrence was seen in patients with unilocular tumours. The Le Fort I osteotomy approach allows direct visualization and ensures wide excision, minimizing the risk of recurrence. In this series, cases with a multilocular KCOT showed a higher risk of recurrence due to the difficulty of removing the tumour in total. All recurrences took place within 2 years of the intervention; a 5-year follow-up is recommended. [ABSTRACT FROM AUTHOR]

Background & Aims Genetic defects in polycystin-1 or -2 ( PC1 or PC2 ) cause polycystic liver disease associated with autosomal dominant polycystic kidney disease (PLD-ADPKD). Progressive cyst growth is sustained by a cAMP-dependent Ras/ERK/HIFα pathway, leading to increased vascular endothelial growth factor A (VEGF-A) signaling. In PC2-defective cholangiocytes, cAMP production in response to [Ca 2+ ] ER depletion is increased, while store-operated Ca 2+ entry (SOCE), intracellular and endoplasmic reticulum [Ca 2+ ] ER levels are reduced. We investigated whether the adenylyl cyclases, AC5 and AC6, which can be inhibited by Ca 2+ , are activated by the ER chaperone STIM1. This would result in cAMP/PKA-dependent Ras/ERK/HIFα pathway activation in PC2-defective cells, in response to [Ca 2+ ] ER depletion. Methods PC2 / AC6 double conditional knockout (KO) mice were generated ( Pkd2 / AC6 KO) and compared to Pkd2 KO mice. The AC5 inhibitor SQ22,536 or AC5 siRNA were used in isolated cholangiocytes while the inhibitor was used in biliary organoid and animals; liver tissues were harvested for histochemical analysis. Results When comparing Pkd2 / AC6 KO to Pkd2 KO mice, no decrease in liver cyst size was found, and cellular cAMP after [Ca 2+ ] ER depletion only decreased by 12%. Conversely, in PC2-defective cells, inhibition of AC5 significantly reduced cAMP production, pERK1/2 expression and VEGF-A secretion. AC5 inhibitors significantly reduced growth of biliary organoids derived from Pkd2 KO and Pkd2 / AC6 KO mice. In vivo treatment with SQ22,536 significantly reduced liver cystic area and cell proliferation in PC2-defective mice. After [Ca 2+ ] ER depletion in PC2-defective cells, STIM1 interacts with AC5 but not with Orai1, the Ca 2+ channel that mediates SOCE. Conclusion [Ca 2+ ] ER depletion in PC2-defective cells activates AC5 and results in stimulation of cAMP/ERK1-2 signaling, VEGF production and cyst growth. This mechanism may represent a novel therapeutic target. Lay summary Polycystic liver diseases are characterized by progressive cyst growth until their complications mandate surgery or liver transplantation. In this manuscript, we demonstrate that inhibiting cell proliferation, which is induced by increased levels of cAMP, may represent a novel therapeutic target to slow the progression of the disease. [ABSTRACT FROM AUTHOR]

Research was conducted to determine the effect of altrenogest and exposure to exogenous gonadotropins on ovarian function in prepubertal and mature gilts. Crossbred, presumably sexually mature gilts (n = 51), were fed with altrenogest for 18 consecutive days and the day after the last feeding with altrenogest, gilts were treated with eCG and 72 hours later challenged with hCG. Animals were slaughtered on Days 10 to 13 of their gonadotropins synchronized estrous cycle. Ovaries were examined for the number of CL, number of follicular cysts, and presence of corpora albicantia. Gilts were divided into two groups: those possessing corpora albicantia (group A—mature; n = 36) and those without corpora albicantia (Group W—prepubertal; n = 15) on their ovaries. In addition, each group was divided into two subgroups depending on the presence of follicular cysts (AC and WC) or their absence (AO and WO). There was no difference between the number of CL in group A and group W. Presence of corpora albicantia determined percentage of gilts possessing follicular cysts (13.9% group A vs. 66.7% group W). Gilts without follicular cysts (AO plus WO; n = 36) had higher number of CL (P < 0.01) than gilts bearing cysts (AC plus WC; n = 15). Comparison AO-AC did not show significant difference (P = 0.075) between CL number in mature cyst-free and cysts bearing gilts. A prepubertal gilts not bearing follicular cysts (WO) had higher (P < 0.02) number of CL than gilts bearing cysts. A significant negative correlation between the number of CL and number of follicular cysts was found (r = −0.664; P = 0.007). There were no differences in blood plasma progesterone and estradiol concentration between cyst-free and cyst-bearing gilts. These results indicate: (1) a higher follicular cysts appearance in prepubertal than mature gilts challenged with altrenogest and exposed to exogenous gonadotropins and (2) a negative effect of follicular cysts on the number of CL (ovulations) in prepubertal gilts. [ABSTRACT FROM AUTHOR]

Autosomal-dominant polycystic kidney disease (ADPKD) is the most commonly inherited kidney disease, and the fourth most common cause of end-stage renal disease. ADPKD is a systemic disorder, associated with numerous extrarenal manifestations, including polycystic liver disease, the most common gastrointestinal manifestation, and diverticular disease, inguinal, and ventral hernias, pancreatic cysts, and large bile duct abnormalities. All of these gastrointestinal manifestations play a significant role in disease burden in ADPKD, particularly in the later decades of life. Thus, as ADPKD becomes more recognized, it is important for gastroenterologists to be knowledgeable of this monogenic disorder’s effects on the digestive system. [ABSTRACT FROM AUTHOR]

This article discusses the common benign cysts found in the vagina and vulva. While not commonly encountered in routine surgical pathology practice, these cysts are a relatively frequent occurrence. The cysts are often asymptomatic and do not come to clinical attention. Cysts of the vagina and vulva can be classified as embryologic or non-embryologic in origin, with müllerian and mesonephric cysts being the most common embryologic cysts. A discussion of the embryologic origin of the vagina and vulva is included as this knowledge can help in accurately characterizing cystic lesions found in this location. Non-embryologic cysts include endometriosis, inclusion cysts, and other rare lesions. These entities are discussed to allow distinction from cysts of embryologic origin. With accurate clinical history, anatomic site of origin, and identification of the epithelial lining, definitive classification of the cyst is possible. [ABSTRACT FROM AUTHOR]

• Mortality rates of planktonic ciliates depend on species and temperature. • Cyst-forming ciliates have higher mortality rates than species that cannot encyst. • Sympatric ciliates may use different strategies to cope with starvation. • Starving mixotrophic species may benefit from resources provided by their endosymbionts. • Mortality rates per day increase with temperature by 0.09 °C−1. We investigated the temperature-dependent response to starvation of three contrasting freshwater ciliates (Ciliophora). The cyst-forming algivorous species Meseres corlissi and the bactivorous species Glaucomides bromelicola , which cannot form cysts, co-occur in the reservoirs (tanks) of tree bromeliads. The mixotrophic species Coleps spetai is common in many lakes. We hypothesized that the ciliates' different traits and life strategies would affect their survival rates and temperature sensitivity under food depleted conditions. We measured the decline of the ciliate populations in microcosm experiments at different temperatures for several days. We used an imaging flow cytometer to size the ciliates and documented their morphological and physiological changes in response to starvation. We found that the cyst-forming species had the highest mortality rates but may endure long-term starvation by encystment. The sympatric, non-encysting species suffered the lowest mortality rates and could survive for more than three weeks without food. The mixotrophic species had intermediate mortality rates but showed the highest phenotypic plasticity in response to starvation. A significant fraction of the C. spetai population appeared unaffected by starvation, suggesting that the endosymbionts provided some resources to the host cells. The mean mortality rate per day of all three species increased with temperature by 0.09 °C−1. [ABSTRACT FROM AUTHOR]

• Rare Lepidodinium eDNA was detected during unfavourable growth seasons. • Increase in Lepidodinium was correlated with ammonium pulse from sediment. • No cysts of Lepidodinium were identified in the sediments. • Temporary pelagic stage during non-blooming phase may explain species overwintering. Green seawater discolorations caused by the marine dinoflagellate Lepidodinium chlorophorum are frequently observed during summer along southern Brittany coasts (NE Atlantic, France). Here, the ecology of L. chlorophorum is studied during a non-bloom period using high-throughput sequencing metabarcoding of environmental DNA (eDNA) samples for the detection of this species at low concentrations. Sediment samples (for metabarcoding and cyst analyses) were collected in January–February 2019 and water samples from two stations were collected at three water depths in September–March 2019–2020 and 2020–2021 (for metabarcoding and environmental parameters). The protistan community was dominated by dinoflagellates and was homogenous in the water column. Amplicon sequence variants (ASVs) associated with the genus Lepidodinium were detected in autumn–winter at low relative abundances (minimum: 0.01%). Increases in Lepidodinium abundance were positively correlated with pulses of ammonium re-suspended from bottom sediments. Although Lepidodinium eDNA (<1%) was detected in the sediments, no cyst morphotypes could be associated with Lepidodinium , and germination experiments revealed no Lepidodinium -like cells, leaving in doubt the existence of resting cysts of this species in the seed bank. It is hypothesised that temporary Lepidodinium cells remained present in the water column at low concentrations during the autumn–winter period, awaiting ammonium input from sediments to initiate growth, and that blooms develop when water column stratification and river input provide favourable environmental conditions for biomass increases. [ABSTRACT FROM AUTHOR]

The mitosome is a double-membrane enveloped organelle that is found in few unicellular eukaryotes, one of which is the human intestinal parasitic protist Giardia intestinalis , which also lacks mitochondria and peroxisomes. This flagellated protist grows in vitro as trophozoites and under some conditions, differentiates into cysts, which are characterized by the absence of externalized flagella, a round shape, and the presence of a cyst wall. The presence and distribution of mitosomal proteins, such as giardial iron-sulfur cluster protein ( GiIscU ), heat-shock protein 70 (mit-HSP70) and giardial chaperonin 60 ( GiCpn60 ), during the process of trophozoite-to-cyst transformation was tracked using confocal laser scanning microscopy and western blotting. During the early stages of the differentiation process (∼12 h), there was a significant decrease in the extent of chaperone labeling in the cells, which disappeared after 21 h but was recovered during the cyst stage; IscU labeling remained present throughout the differentiation process. This finding was confirmed by mRNA expression analysis, thus indicating that a process modulates the expression of mitosomal chaperones during the G . intestinalis life cycle. Microscopy techniques, such as structured illumination and electron tomography, revealed a novel profile for central mitosomes, as well as the presence of both rounded and elongated mitosomes. [ABSTRACT FROM AUTHOR]

Retroperitoneal cystic mesothelioma is a very rare lesion. The pathogeny is unclear and establishing a preoperative diagnosis versus others retroperitoneal cystic lesions is difficult. Thus, with increasing experience in laparoscopic retroperitoneal surgery, the use of this approach for exploration of a retroperitoneal mass of unknown origin may provide an alternative to classic open surgery and all the benefits of laparoscopy. We present two cases treated laparoscopycally and review the literature. [ABSTRACT FROM AUTHOR]

Pulmonary hyalinizing clear cell carcinoma (HCCC) is a new and rare form of lung salivary gland tumor. Only twenty-two cases have been reported in the literature to date. Furthermore, their clinicopathological features have not been fully characterized. In this paper, we describe the clinicopathological characteristics, immunohistochemical features, and molecular genetic changes in two HCCC cases. We also simultaneously reviewed related literature on similar cases reported. Of the two cases, one was of a 58-year-old man with a 4.3 cm lung tumor, which was the largest among all previously reported cases. The tumor showed an infiltrative growth pattern and perineural and vascular invasion microscopically. Moreover, nuclear grooves, high mitotic figures, and comedo necrosis were observed in addition to classic morphological features. More importantly, rare pseudopapillary structures were observed. The second case was of a 60-year-old woman in whom the tumor was mainly composed of multiple cysts filled with mucus. The remaining focal solid areas of the tumor comprised clear and acidophilic cells embedded in the hyalinizing stroma. Immunohistochemical analysis revealed that the tumor cells of both cases were positive for CK5/6, p40, and p63 expression, but negative for napsin A, TTF-1, and SOX10 expression. The HCCC diagnosis in both cases was validated by fluorescence in-situ hybridization (FISH) examination, which showed Ewing sarcoma breakpoint region 1-activating transcription factor 1 (EWSR1-ATF1) gene fusion. Primary pulmonary HCCC is a rare lung tumor originating from the bronchial mucosa, and its histological features may vary, such as rare pseudopapillary structures and abundant cysts. Thus, the diagnosis should be a combined analysis of histopathological characteristics with immunophenotype and molecular examination, including EWSR1-ATF1 gene fusion detection. [ABSTRACT FROM AUTHOR]

The acritarch genus Radiosperma has been reported from plankton and sediments since the late 19th century, with suggested biological affinities ranging from invertebrate eggs to tintinnids. Here, the genus description is improved and its two species, Radiosperma corbiferum and Radiosperma textum , are redescribed. Radiosperma textum is shown to be a ciliate cyst related to Askenasia based on new SSU and LSU rRNA sequences. The spatiotemporal distribution and ecology of both species are discussed. The chemical composition is documented based on micro-Fourier Transform Infrared spectroscopy. Furthermore, new SSU and LSU rRNA sequences for several flask shaped ciliate cysts (Fusopsis , Strombidium) are also included in the analysis and the occurrence of fossilizable cysts in the ciliophoran clade is reviewed. • Former acritarch Radiosperma and its two species R. corbiferum and R. textum are redescribed as ciliates. • Radiosperma corbiferum apears confined to Baltic and Arctic waters while R. textum occurs in temperate coastal waters. • Radiosperma textum is a ciliate cyst based on rRNA. • Ciliate cyst morphology has taxonomic significance. [ABSTRACT FROM AUTHOR]

A new benthic toxic dinoflagellate is described from the Celebes Sea. Gambierdiscus balechii sp. nov. was isolated from seaweeds growing in tidal ponds. Its morphology was studied by means of LM and SEM; G. balechii has a very ornamented theca, a hatchet shaped second apical plate, a narrow second antapical plate and an asymmetrical third precigular plate, a unique combination of characters among Gambierdiscus species. It has a very wide size range with widths from 36 to 88 μm. Phylogenetic analyses of two G. balechii strains, based on LSU rRNA (D8–D10) and partial SSUrRNA sequences confirmed that these clustererd in its’ own group, separated from the rest of Gambierdiscus species and with G. pacificus , G. belizeanus and G. scabrosus as its closest relatives. Thecate cysts are described from culture as non motile vegetative-like cells which germinated after being isolated and transferred to fresh medium. Mouse tests showed that this species is toxic and hence it is a potential cause of ciguatera in the Celebes Sea. [ABSTRACT FROM AUTHOR]

The development of perifocal edema and tumor bed cyst has been reported after implantation of biodegradable carmustine wafers for the treatment of malignant gliomas. We retrospectively evaluated these changes in a series of patients; 19 consecutive patients with malignant glioma who received carmustine wafer implantation at our hospital from January 2013 through July 2013, and 28 patients who underwent surgery prior to our institution’s initiation of carmustine wafer implantation, as historical controls. The volume of the tumor bed cyst and perifocal edema was calculated on MRI acquired at four time points: ⩽72 hours after surgery for baseline, and at 1–4, 5–8, and 9–12 weeks after surgery. The volume of the tumor bed cyst in the wafer group increased significantly relative to the control group at all time points ( p = 0.04). Opening of the ventricle was inversely correlated with enlargement of the tumor bed cyst in the wafer group ( p = 0.04). The change in the volume of perifocal edema in the wafer group was not significantly different ( p = 0.48), but exhibited a considerable increase in patients with anaplastic oligodendroglioma relative to glioblastoma patients in the wafer group ( p = 0.01). We demonstrated significant enlargement of the tumor bed cyst volume after carmustine wafer implantation, as well as the development of marked perifocal edema in patients with anaplastic oligodendroglioma. [ABSTRACT FROM AUTHOR]

Toxoplasma gondii is a ubiquitous protozoan parasite with approximately one-third of the worlds' population chronically infected. In chronically infected individuals, the parasite resides primarily in cysts within neurons in the central nervous system. The chronic infection in immunocompetent individuals has been considered to be asymptomatic but increasing evidence indicates the chronic infection can lead to neuropsychiatric disorders such as Schizophrenia, prenatal depression and suicidal thoughts. A better understanding of the mechanism(s) by which the parasite exerts effects on human behavior is limited due to lack of suitable human neuronal models. In this paper, we report the use of human neurons derived from normal cord blood CD34+ cells generated via genetic reprogramming, as an in vitro model for the study T. gondii in neurons. This culture method resulted in a relatively pure monolayer of induced human neuronal-like cells that stained positive for neuronal markers, MAP2, NFL, NFH and NeuN. These induced human neuronal-like cells (iHNs) were efficiently infected by the Prugniad strain of the parasite and supported replication of the tachyzoite stage and development of the cyst stage. Infected iHNs could be maintained through 5 days of infection, allowing for formation of large cysts. This induced human neuronal model represents a novel culture method to study both tachyzoite and bradyzoite stages of T. gondii in human neurons. [ABSTRACT FROM AUTHOR]

Introduction Ciliated foregut cysts (CFC) are rare anomalies due to aberrant embryological development. It is thought to arise from a remnant of the embryologic foregut. The solitary cysts are characterised by ciliated pseudostratified columnar epithelium. They are usually located above the diaphragm but they can also arise in relation to the liver, gallbladder and pancreas. Presentation of case We present the first ciliated foregut cyst of the gallbladder case reported in Australia, and the ninth known case to be reported worldwide. A 61-year-old male with chronic cholecystitis and cholelithiasis underwent an elective laparoscopic cholecystectomy and intraoperative cholangiogram. Intraoperatively, ‘out-pouching’ was noted on the lateral border of the gallbladder. Microscopically the histopathology showed that the cyst was lined by ciliated columnar epithelium the characteristic feature of a ciliated foregut cyst. Discussion To date only 8 cases of these ciliated foregut cysts in the gallbladder have been reported in literature. Our case is the first reported in Australia. It is unique in that the patient was an older male as opposed to most other previous cases, which were younger females. These cysts can be difficult to distinguish from neoplasms clinically and radiographically. Reports have shown that these cysts may become dysplastic and is best excised when discovered. Conclusion Despite the rarity of CFCs and their potential to mimic malignancy, we propose awareness and understanding of the management for them—being excision and hopefully not cause any confusion or devastatingly allow it to become malignant. [ABSTRACT FROM AUTHOR]