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181. PKD1-associated autosomal dominant polycystic kidney disease with glomerular cysts presenting with nephrotic syndrome caused by focal segmental glomerulosclerosis. (PubMed)

PKD1-associated autosomal dominant polycystic kidney disease with glomerular cysts presenting with nephrotic syndrome caused by focal segmental glomerulosclerosis. Autosomal dominant polycystic kidney disease (ADPKD) may manifest non-nephrotic range proteinuria, but is rarely complicated with nephrotic syndrome. Limited number of reports describe the histology of ADPKD with nephrotic syndrome in detail.We encountered a 23-year-old man with polycystic kidney disease (PKD) with small kidney (...) volume and nephrotic syndrome, which eventually progressed to end-stage renal disease. Renal histology showed typical focal segmental glomerulosclerosis and remarkable glomerular cyst formation, but did not reveal tubular cysts. PKD1 mutation was detected in him and his father, who also had PKD with small kidney volume.In contrast to tubular cysts which develop along ADPKD progression, glomerular cysts may likely be associated with ADPKD with slower volume progression manifesting small kidney volume

2019 BMC Nephrology

182. Prenatal diagnosis of HNF1B-associated renal cysts: Need to differentiate intragenic variants from 17q 12 microdeletion syndrome? (PubMed)

Prenatal diagnosis of HNF1B-associated renal cysts: Need to differentiate intragenic variants from 17q 12 microdeletion syndrome? 17q12 microdeletions containing HNF1B and intragenic variants within this gene are associated with variable developmental, endocrine and renal anomalies, often already noted prenatally as hyperechogenic/cystic kidneys. Here, we describe pre- and postnatal phenotypes of seven individuals with HNF1B aberrations and compare their clinical and genetic data to previous

2019 Prenatal diagnosis

183. Giant epidermoid cyst of the spleen in a pediatric patient: A case report. (PubMed)

Giant epidermoid cyst of the spleen in a pediatric patient: A case report. Splenic cysts (SCs) are rare findings in children, particularly the youngest. Here, we discuss a case that is useful for the differential diagnosis and treatment of SCs.A 9-year-old Albanian boy was admitted for severe abdominal pain localized mainly in the left hypochondrium for approximately 24 hours. His medical history was without significant clinical problems.Splenomegaly was diagnosed during the first clinical

2019 Medicine

184. Nonsurgical integrative Korean Medicine treatment of discal cyst: A case report and a retrospective chart review analysis. (PubMed)

Nonsurgical integrative Korean Medicine treatment of discal cyst: A case report and a retrospective chart review analysis. Discal cysts are rare lesions characterized by pain caused by neurogenic compression with similar symptoms as those of disc herniation. This study aims to report the spontaneous regression of discal cyst achieved through nonsurgical integrative Korean Medicine treatment and the clinical epidemiological features of discal cyst cases collected from 4 institutions.A 31-year (...) -old woman had low back pain and radiating pain equivalent to a numeral rating scale (NRS) of 8 and had limitations in daily work and activities.The patient was diagnosed as having discal cysts that compressed the left S1 based on findings of L-spine magnetic resonance imaging (MRI) performed at our hospital.The patient received nonsurgical Korean Medicine treatment and after 24 days of treatment in the hospital, she underwent 16 additional treatments as an outpatient.Spontaneous regression

2019 Medicine

185. Esophageal duplication cyst presenting with stridor in a child with congenital pulmonary airway malformation: A case report and literature review. (PubMed)

Esophageal duplication cyst presenting with stridor in a child with congenital pulmonary airway malformation: A case report and literature review. Esophageal duplication cyst (EDC) is a rare developmental aberration originated from the embryonic foregut. It may remain asymptomatic but produce local mass effect on surrounding organs if rapid enlarges. EDC may sometimes accompany with other congenital malformations. Congenital pulmonary airway malformation (CPAM) is a congenital lung malformation (...) with an unknown chance of developing symptoms. Here we report a rare case of esophageal duplication cyst with type 2 congenital pulmonary airway malformation (CPAM).A 16-month old boy with a prenatal diagnosis of type 2 CPAM presented progressive stridor and respiratory distress and was admitted to our hospital under the diagnosis of pneumonia. The patient responded poorly to antibiotics. A chest Xray (CXR) showed consolidation over the left upper lobe with trachea deviated to right side. A chest computed

2019 Medicine

186. Fetal giant right cervical cyst causing severe tracheal compression: A case report. (PubMed)

Fetal giant right cervical cyst causing severe tracheal compression: A case report. Fetal giant cervical cyst (FGCC) is a rare congenital anomaly. Sometimes FGCC may extend into the mediastinum, and result in severe tracheal compression, which is a life-threatening event at birth.We present a rare case of FGCC, which extended from the right neck into the superior mediastinum, and resulted in severe tracheal compression.An FGCC was observed by ultrasonography and magnetic resonance imaging (MRI (...) ) at 27+4 weeks' gestation (WG). Fetal MRI at 35+1 WG showed that the FGCC was 3.3 × 8.2 × 7.5 cm and extended from the right neck into the superior mediastinum. Severe tracheal compression was observed and the inside diameter of the narrowest section of tracheostenosis appeared thread-like and measured only 0.1 cm.Cervical cyst reduction was performed prenatally under ultrasound guidance to alleviate the tracheal compression and maximize the chance of fetal survival 2 days before birth. At 36+3 WG

2019 Medicine

187. A suprasellar bronchogenic cyst: A case report. (PubMed)

A suprasellar bronchogenic cyst: A case report. Bronchogenic cysts are mainly benign, congenital abnormalities, originating from the remnants of the primitive foregut. However, intracranial bronchogenic cysts have been rarely observed. Hence, better understanding of the suprasellar bronchogenic cysts is of great significance to properly perform perioperative management.A 62-year-old Chinese female was admitted to our hospital due to impairment of memory and asthenia.Magnetic resonance imaging (...) (MRI) confirmed presence of cystic lesion in the suprasellar region. The patient underwent craniotomy with resection of the cystic mass. The histopathological examinations confirmed diagnosis of bronchogenic cyst as well.The cystic lesion was removed without complications. No drugs during follow-up were recommended.After discharge, the patient was advised to undergo MRI for 6 months to 1 year. No evidence of recurrence was found in the first postoperative review.Bronchogenic cysts should

2019 Medicine

188. Correction to: A child with bilateral multiple renal cysts presenting with ascites and pleural effusion: answers. (PubMed)

Correction to: A child with bilateral multiple renal cysts presenting with ascites and pleural effusion: answers. The original version of this article unfortunately contained a mistake. The answer to question 3 was presented incorrectly. The correct answer is given below.

2019 Pediatric Nephrology

189. Complete excision of esophageal bronchogenic cyst by endoscopic submucosal tunnel dissection: a case presentation. (PubMed)

Complete excision of esophageal bronchogenic cyst by endoscopic submucosal tunnel dissection: a case presentation. Intramural esophageal bronchogenic cyst is very rare. Surgical removal of the cysts is advised even the patients are asymptomatic, since the cyst can lead to complications, and there is a risk of malignant transformation. Thoracotomy or thoracoscopy is the most commonly used approach for complete excision of the cysts. To our knowledge, this is the first report to excise intramural (...) esophageal bronchogenic cyst completely by endoscopic submucosal tunnel dissection (ESTD).A 40-year-old male was referred to our hospital due to the detection of a submucosal tumor at the distal esophagus. The tumor was found during gastroendoscopy in a general health check-up. The patient had no symptoms. A benign esophageal tumor was confirmed by endoscopic ultrasonography (EUS) and computed tomography (CT). On the basis of these results, ESTD was performed. During the procedure, a cystic mass

2019 BMC Gastroenterology

190. Follow-up of Incidentally Detected Pancreatic Cystic Neoplasms: Do Baseline MRI and CT Features Predict Cyst Growth? (PubMed)

Follow-up of Incidentally Detected Pancreatic Cystic Neoplasms: Do Baseline MRI and CT Features Predict Cyst Growth? Background Incidental detection of pancreatic cystic neoplasm (PCN) has increased. Since a small percentage of PCNs possess malignant potential, management is challenging. The recently revised American College of Radiology (ACR) recommendations define PCN measurement and growth for different categories based on baseline cyst size. However, no data are available regarding PCN (...) recommendations. Cyst features, including location, septations, and mural nodules and multiplicity, were noted. Time to cyst progression (growth by ACR criteria) was examined by using baseline PCN size, among other factors. Results A total of 646 cysts in 390 patients were followed up for a median of 50 months (range, 12-186 months). A total of 184 (28.5%) cysts increased in size, 52 (8.1%) decreased in size, and 410 (63.4%) remained stable. For groups in which baseline PCN size was smaller than 5 mm, 5-14 mm

2019 Radiology

191. Simple Adnexal Cysts: SRU Consensus Conference Update on Follow-up and Reporting. (PubMed)

Simple Adnexal Cysts: SRU Consensus Conference Update on Follow-up and Reporting. This multidisciplinary consensus update aligns prior Society of Radiologists in Ultrasound (SRU) guidelines on simple adnexal cysts with recent large studies showing exceptionally low risk of cancer associated with simple adnexal cysts. Most small simple cysts do not require follow-up. For larger simple cysts or less well-characterized cysts, follow-up or second opinion US help to ensure that solid elements (...) are not missed and are also useful for assessing growth of benign tumors. In postmenopausal women, reporting of simple cysts greater than 1 cm should be done to document their presence in the medical record, but such findings are common and follow-up is recommended only for simple cysts greater than 3-5 cm, with the higher 5-cm threshold reserved for simple cysts with excellent imaging characterization and documentation. For simple cysts in premenopausal women, these thresholds are 3 cm for reporting

2019 Radiology

192. PLCD1 and Pilar Cysts. (PubMed)

PLCD1 and Pilar Cysts. Trichilemmal or "pilar" cysts are commonly found on the scalp and are derived from the outer root sheath of the hair follicle. Multiple trichilemmal cysts present in an autosomal dominant pattern of inheritance, yet the genetic mechanism has remained elusive. In this issue, Hörer et al. (2019) highlight predisposing variants in PLCD1 in such families and propose a monoallelic mutational mechanism that drives cyst formation.Copyright © 2019 The Authors. Published

2019 Journal of Investigative Dermatology

193. Case Report: Laparoscopic Excision of a Primary Giant Splenic Hydatid Cyst: Literature Review. (PubMed)

Case Report: Laparoscopic Excision of a Primary Giant Splenic Hydatid Cyst: Literature Review. Cystic echinococcosis (CE), also known as hydatid cyst, is a zoonosis caused by the tapeworm Echinococcus granulosus. It is a common health problem in many countries. This condition predominantly affects the liver and the lungs, and the spleen to a less extent (splenic hydatid cyst, SHD). Indeed, it is estimated that SHD occurs in less than 2% of abdominal CE and 0.5-8% of CE cases. Here, we present (...) . In addition to this case, we review the previous literature on SHD treated by laparoscopy and reveal that laparoscopic approach is safe and effective for SHD. Particularly, we show that laparoscopic splenectomy is feasible for giant cysts (> 10 cm) at high risk of rupture or compressing other vital structures.

2019 American Journal of Tropical Medicine & Hygiene

194. A longitudinal analysis of albendazole treatment effect on neurocysticercosis cyst evolution using multistate models. (PubMed)

A longitudinal analysis of albendazole treatment effect on neurocysticercosis cyst evolution using multistate models. In neurocysticercosis, the larval form of the pork tapeworm Taenia solium appears to evolve through three phases-active, degenerative and sometimes calcification-before disappearance. The antihelmintic drug, albendazole, has been shown to hasten the resolution of active cysts in neurocysticercosis. Little is known about the time cysts take to progress through each phase (...) , with or without treatment.We reconfigured brain imaging data from patient level to cyst level for 117 patients in a randomized clinical trial of albendazole in which images were taken at baseline, 1, 6, 12 and 24 mo. Applying a multistate model, we modelled the hazard of a cyst evolving to subsequent cyst phases before the next imaging (vs no change). We examined the impact of albendazole treatment overall and by patient and cyst characteristics on the hazard.Albendazole accelerated the evolution from

2019 Transactions of the Royal Society of Tropical Medicine & Hygiene

195. The combination of Cytokines and albendazole therapy for prophylaxis and treatment of experimental ./hydatid cyst. (PubMed)

The combination of Cytokines and albendazole therapy for prophylaxis and treatment of experimental ./hydatid cyst. Albendazole is appropriate chemotherapy for treatment and prophylaxis of cystic echinococcosis (CE). The recent studies show Cytokine therapy could be useful for chronic and progressive diseases, therefore, the use of cytokine in prophylaxis and treatment of hydatidosis could be considerable. The aim of this study is to evaluate the efficacy of interleukin-12 + interferon-gamma (...) , in combination with albendazole for prophylaxis and treatment of CE in Balb/c mice. Albendazole and cytokines were administrated for prophylaxis and treatment in CE. The efficacy of this agents were determined by measuring of size, weight, number of cysts, histology and lymphocyte response. Lymphocyte stimulation index and production of interleukin-12 and interferon-gamma were measured by MTT assay and ELISA respectively. Combination of albendazole and cytokines was very effective for prophylaxis

2019 Acta Tropica

196. Cell-Autonomous Hedgehog Signaling Is Not Required for Cyst Formation in Autosomal Dominant Polycystic Kidney Disease. (PubMed)

Cell-Autonomous Hedgehog Signaling Is Not Required for Cyst Formation in Autosomal Dominant Polycystic Kidney Disease. PKD1 or PKD2, the two main causal genes for autosomal dominant polycystic kidney disease (ADPKD), encode the multipass transmembrane proteins polycystin-1 (PC1) and polycystin-2 (PC2), respectively. Polycystins localize to the primary cilium, an organelle essential for cell signaling, including signal transduction of the Hedgehog pathway. Mutations in ciliary genes that build (...) and maintain the cilium also cause renal cystic disease through unknown pathways. Although recent studies have found alterations in Hedgehog signaling in ADPKD-related models and tissues, the relationship between Hedgehog and polycystic kidney disease is not known.To examine the potential role of cell-autonomous Hedgehog signaling in regulating kidney cyst formation in vivo in both early- and adult-onset mouse models of ADPKD, we used conditional inactivation of Pkd1 combined with conditional modulation

2019 Journal of the American Society of Nephrology

197. <i>ALG9</i> Mutation Carriers Develop Kidney and Liver Cysts. (PubMed)

ALG9 Mutation Carriers Develop Kidney and Liver Cysts. Mutations in PKD1 or PKD2 cause typical autosomal dominant polycystic kidney disease (ADPKD), the most common monogenic kidney disease. Dominantly inherited polycystic kidney and liver diseases on the ADPKD spectrum are also caused by mutations in at least six other genes required for protein biogenesis in the endoplasmic reticulum, the loss of which results in defective production of the PKD1 gene product, the membrane protein (...) after age 50; seven (88%) had at least four kidney cysts, compared with none in matched controls without ALG9 mutations.ALG9 is a novel disease gene in the genetically heterogeneous ADPKD spectrum. This study supports the utility of phenotype characterization in genetically-defined cohorts to validate novel disease genes, and provide much-needed genotype-phenotype correlations.Copyright © 2019 by the American Society of Nephrology.

2019 Journal of the American Society of Nephrology

198. Timing of choledochal cyst perforation. (PubMed)

Timing of choledochal cyst perforation. Choledochal cyst (CDC) perforation with bile peritonitis is potentially fatal. This study aims to establish the optimal timing for a CDC operation. Data of 133 children with perforated CDCs managed at our institution between August 2011 and July 2017 were reviewed. The perforations were categorized: Type 1: Complete cyst perforation with ascites and general peritonitis (n=52), Type 2: Sealed perforation with a small amount of bile contained

2019 Hepatology

199. "Trivial" Cysts Redefine the Risk of Cancer in Presumed Branch-Duct Intraductal Papillary Mucinous Neoplasms of the Pancreas: A Potential Target for Follow-Up Discontinuation? (PubMed)

"Trivial" Cysts Redefine the Risk of Cancer in Presumed Branch-Duct Intraductal Papillary Mucinous Neoplasms of the Pancreas: A Potential Target for Follow-Up Discontinuation? The management of small and incidental branch duct intraductal papillary mucinous neoplasms (BD-IPMNs) still is of concern. The aim is assessing the safety of a surveillance protocol through the evaluation of their progression to malignancy.All presumed BD-IPMNs observed from 2000 to 2016 were included. Only patients (...) a median of 62 months. The median cyst growth rate was 0 mm/yr. A growth rate ≥2.5 mm/yr and the development of WF resulted independent predictors of PC. The standardized incidence ratio of PC for trivial BD-IPMN (n = 378) was 22.45 (95% confidence interval 8.19-48.86), but considering only patients aged >65 years (n = 198), it decreased to 3.84 (95% confidence interval 0.77-11.20).Surveillance of the vast majority of presumed BD-IPMNs is safe, as the risk of PC is comparable to postoperative mortality

2019 American Journal of Gastroenterology

200. An Unusual Cause of Pulmonary Arterial Hypertension: Hydatid Cyst. (PubMed)

An Unusual Cause of Pulmonary Arterial Hypertension: Hydatid Cyst. Hydatid cyst is an important health problem throughout the world and it is caused by the larval form of Echinococcus granulosus. Although it is most commonly located in the liver and lungs, hydatid cyst can also affect other organs because the oncospheres spread through the bloodstream. The disease may have many different clinical presentations. Although it is a benign disease, sometimes it can cause serious morbidity and even (...) mortality. Here, the case of a 33-year-old male patient who underwent surgery for a right ventricle and bilateral lung hydatid cyst 9 years ago and had pulmonary hypertension is presented.Copyright © 2019. Published by Elsevier Inc.

2019 Annals of Thoracic Surgery

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