Search

Autosomal dominant polycystic kidney disease (ADPKD) is caused by mutations in PKD1 and PKD2, encoding polycystin-1 (PC1) and polycystin-2 (PC2), which are required for the regulation of the renal tubular diameter. Loss of polycystin function results in cyst formation. Atypical forms of ADPKD are caused by mutations in genes encoding endoplasmic reticulum (ER)-resident proteins through mechanisms that are not well understood. Here, we investigate the function of DNAJB11, an ER co-chaperone associated with atypical ADPKD. We generated mouse models with constitutive and conditional Dnajb11 inactivation and Dnajb11-deficient renal epithelial cells to investigate the mechanism underlying autosomal dominant inheritance, the specific cell types driving cyst formation, and molecular mechanisms underlying DNAJB11-dependent polycystic kidney disease. We show that biallelic loss of Dnajb11 causes cystic kidney disease and fibrosis, mirroring human disease characteristics. In contrast to classical ADPKD, cysts predominantly originate from proximal tubules. Cyst formation begins in utero and the timing of Dnajb11 inactivation strongly influences disease severity. Furthermore, we identify impaired PC1 cleavage as a potential mechanism underlying DNAJB11-dependent cyst formation. Proteomic analysis of Dnajb11- and Pkd1-deficient cells reveals common and distinct pathways and dysregulated proteins, providing a foundation to better understand phenotypic differences between different forms of ADPKD., (© 2024 The Author(s). The FASEB Journal published by Wiley Periodicals LLC on behalf of Federation of American Societies for Experimental Biology.)

Aim: Due to the rarity of lung cancer with cystic imaging manifestations, we explore the clinical features and survival prognosis of such tumors., Materials and Methods: Imaging characteristics were used to categorize 3,556 patients who underwent surgery for isolated primary lung cancer into one of three groups: those with cystic lung cancer (149), solid lung cancer (1,399), and ground-glass lung cancer (1,160). Propensity score matching by sex and age was performed to analyze the differences in clinical characteristics of lung cancer among the three groups and the correlation between clinical characteristics of cystic lesions and progression-free survival (PFS)., Results: The three groups of patients differed in various aspects, including pathological type, smoking history, tumor stage, type of surgery, histological grading, and PFS (P < 0.05). The results of the multifactorial analysis indicated that lung cancer type, pathological type, lymph node metastasis, tumor stage, and histologic grading were independent prognostic factors for lung cancer (P < 0.05). After comparison, there was a difference in prognosis between cystic lung cancer and ground-glass lung cancer (P < 0.05)., Conclusion: The clinical features of cystic lung cancer are significantly different from those of ground-glass lung cancer and solid lung cancer. Cystic lesions are independent influencing factors affecting lung cancer, and the prognosis of cystic lung cancer is worse than that of ground-glass lung cancer., (Copyright © 2024 The Royal College of Radiologists. Published by Elsevier Ltd. All rights reserved.)

A 37-year-old man with autosomal dominant polycystic kidney disease (ADPKD) was admitted to our hospital with a liver volume of 8,000 cm 3 . Hepatic arterial embolization was performed using a microcoil but was ineffective. Eight years later, the hepatomegaly progressed to liver failure and death. At autopsy, the liver weighed 21.5 kg, and the entire liver had been replaced by cysts; in the few remaining areas of liver parenchyma, microscopic, small cysts of various sizes and fibrosis were evident, with only a few normal hepatocytes observed. Hepatic arterial branches developed; however, the portal vein could not be observed.

The figure shows tissue samples taken from three previous cases, revealing the cause of hemosiderin deposition in the central nervous system because of superficial siderosis., (© 2024 The Authors. Brain Pathology published by John Wiley & Sons Ltd on behalf of International Society of Neuropathology.)

Abstract: Cutaneous ciliated cysts are rare benign lesions most commonly seen in the lower extremities of young female patients. There is still ongoing debate about the origin of these cysts, with the most common theories being Mullerian heterotopia and eccrine gland metaplasia. To further increase the data pool, we present 2 new cutaneous ciliated cyst cases and gathered data about all 67 cases of literature. We want to highlight that immunohistochemistry and demographic information may play a key role in distinguishing between the 2 possible origins of this cyst, namely estrogen receptor, progesterone receptor, and carcinoembryonic antigen immunohistochemistry. Estrogen receptor and progesterone receptor nuclear positivity is proof of Mullerian origin, but when they are negative with a positive carcinoembryonic antigen, the most likely origin is eccrine gland metaplasia. We advocate using the terms "Cutaneous Mullerian Cyst" and "Ciliated Cutaneous Eccrine Cyst" instead of "Cutaneous Ciliated Cyst" to accurately indicate their distinct origins., Competing Interests: The authors declare no conflicts of interest., (Copyright © 2024 Wolters Kluwer Health, Inc. All rights reserved.)

Background: Autosomal Dominant Polycystic Kidney Disease (ADPKD) is a prevalent genetic disorder characterized by the formation of renal cysts leading to kidney failure. Despite known genetic underpinnings, the variability in disease progression suggests additional regulatory layers, including epigenetic modifications., Methods: We utilized various ADPKD models, including Pkd1 and Ezh2 conditional knockout (Pkd1 delta/delta :Ezh2 delta/delta ) mice, to explore the role of Enhancer of Zeste Homolog 2 (EZH2) in cystogenesis. Pharmacological inhibition of EZH2 was performed using GSK126 or EPZ-6438 across multiple models., Results: EZH2 expression was significantly upregulated in Pkd1 -/- cells, Pkd1 delta/delta mice, and human ADPKD kidneys. EZH2 inhibition attenuates cyst development in MDCK cells and a mouse embryonic kidney cyst model. Both Ezh2 conditional knockout and GSK126 treatment suppressed renal cyst growth and protected renal function in Pkd1 delta/delta mice. Mechanistically, cAMP/PKA/CREB pathway increased EZH2 expression. EZH2 mediated cystogenesis by enhancing methylation and activation of STAT3, promoting cell cycle through p21 suppression, and stimulating non-phosphorylated β-catenin in Wnt signaling pathway. Additionally, EZH2 enhanced ferroptosis by inhibiting SLC7A11 and GPX4 in ADPKD., Conclusion: Our findings elucidate the pivotal role of EZH2 in promoting renal cyst growth through epigenetic mechanisms and suggest that EZH2 inhibition or ablation may serve as a novel therapeutic approach for managing ADPKD., (© 2024. The Author(s).)

Objective: To describe the clinicopathological features of 3 new cases of soft-tissue keratocyst (STK) of the buccal mucosa from Brazil., Materials and Methods: Clinical data from 3 patients diagnosed with STK were obtained from the archives of the Laboratory of Oral Pathology at the Federal University of Rio de Janeiro- Brazil from 2020 to 2023. Two oral pathologists reviewed conventional hematoxylin and eosin-stained slides of each case. Immunohistochemical stainings for CK19, Bcl-2, CD138, D2-40, EMA, and Ki-67 were performed in all cases., Results: Case 1 was a 58-year-old man with a 2-year history of painless swelling of the right buccal mucosa, measuring approximately 5 cm. Case 2 was of a 44-year-old man with a painful swelling in the left buccal mucosa lasting 3 years with drainage to the oral cavity. Case 3 was of a 74-year-old woman with a painful swelling in the left buccal mucosa of unknown duration. Microscopic evaluation of all 3 cases revealed a cyst lined with a few cell layers composed of columnar palisading basal cells and a corrugated parakeratin surface. The capsule comprised fibrous connective tissue with variable amounts of blood vessels with hemorrhage, nerve bundles, fat tissue, striated muscle fibers, and the absence of pilosebaceous units. No recurrence was observed after complete surgical removal. All epithelial layers of the 3 cases expressed positivity for CK19 and CD138, the basal cells were positive for Bcl-2 and D2-40, and the superficial epithelial layer was positive for EMA. All cases demonstrated a low proliferation index by Ki-67., Conclusion: This study represents a series of 3 cases of STK that affected the posterior buccal mucosa of middle-aged patients from Brazil, sharing histopathological and immunohistochemical features with odontogenic keratocyst., (© 2024. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Retropharyngeal goitre extending to the oropharyngeal level is rare. We present a case of papillary thyroid carcinoma (PTC) with a retropharyngeal cystic goitre extending to the uvular level. A woman in her 50s presented with swelling of the neck and dyspnoea. CT and MRI findings showed a primary tumour in the left lobe of the thyroid gland and a retropharyngeal dumbbell-shaped cystic goitre extending to the uvular level. Total thyroidectomy, central neck dissection and tracheostomy were performed. During the surgery, we opened the retropharyngeal space, and no mass was found. The pathological findings showed that the primary PTC (pT2) was surrounded by benign lesions, including the dumbbell-shaped cystic goitre. We speculated that the dumbbell-shaped cystic goitre extended from the visceral space (VS) into the pharyngeal mucosal space (PMS) and reached the uvular level because the thyroid gland is in the VS, and the VS and PMS are continuous spaces., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

Abstract: A 23-year-old man with papillary thyroid cancer underwent radioiodine ( 131 I) treatment following total thyroidectomy and node dissection. Posttreatment 131 I SPECT/CT images revealed an additional focus of increased 131 I activity in the left adrenal gland, raising suspicion for possible adrenal metastasis. Ultimately, pathological examination confirmed the diagnosis of an adrenal cyst., Competing Interests: Conflicts of interest and sources of funding: none declared., (Copyright © 2024 Wolters Kluwer Health, Inc. All rights reserved.)

Rectal implantation cysts can occur at anastomotic sites after low anterior resection (LAR) for rectal cancer. Herein, we report a case of primary adenocarcinoma arising from a rectal implantation cyst after LAR for rectal cancer. A 70-year-old woman was referred to our hospital for diagnosis and treatment of a growing cystic lesion. She had LAR performed for rectal cancer 29 years previously and had a rectal implantation cyst detected 13 years previously. On the first visit to our hospital, serum CEA and CA19-9 levels were elevated, and computed tomography (CT) scans revealed a cystic lesion near the anastomosis. CT-guided biopsy revealed no cancer tissue in the cystic lesion. After that, the cystic lesion naturally shrank, and serum CEA and CA19-9 levels became normal. Follow-up included 3 monthly serum CEA and CA19-9 testing and 6 monthly CT scans. Two years later, serum CEA and CA19-9 levels were elevated again. Colonoscopy revealed an ulcerative lesion at the anastomotic site, in which adenocarcinoma was confirmed. Abdominoperineal resection with sacral resection was performed, and postoperative histopathological examination revealed a primary adenocarcinoma with mucinous component at the implantation cyst. Since rectal implantation cysts can become malignant after extended periods, clinicians need to be aware of this disease., (© 2024. Japanese Society of Gastroenterology.)

Objectives: We aimed to investigate the relationship between the presence of the accessory maxillary ostium (AMO) with mucosal thickening (MT) and maxillary sinus cysts (MSCs)., Study Design: This single-center retrospective study included patients who underwent paranasal sinus computed tomography (CT) between November 2022 and February 2023. We excluded patients with natural maxillary ostium (NMO) occlusion. Two radiologists evaluated the presence of AMOs, MT, and MSCs in CT images and measured their dimensions using a three-dimensional volumetric analysis program., Results: We included 125 patients (mean age: 36.47 ± 16.0 years; male/female: 67/58). In total, 65 patients (52%) had single or multiple AMOs. No significant relationship was found between the presence of an AMO and the presence of MT on either side (P ≥ .306). There was a statistically significant difference between the presence of an AMO and MSC on the right side (P = .002), but not on the left side (P = .477). The diameter of the AMO and the distance from the AMO to the maxillary sinus floor were not correlated with mucosal thickness or MSC volume, but the distance from the AMO to the sinus floor had a small positive correlation with total maxillary sinus volume., Conclusions: The presence of an AMO and its increased diameter may not contribute to ipsilateral maxillary sinusitis in patients whose unilateral or bilateral NMOs are not occluded., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Background: Tuberculous peritonitis often presents with nonspecific symptoms that can lead to diagnostic challenges, particularly when manifesting as peritoneal pseudocysts. This study highlights the clinical complexity and diagnostic approach of tuberculous peritonitis presented as a pseudocyst in an immunocompetent adult, an atypical scenario that is rarely documented., Case Presentation: We report a detailed case of a 41-year-old man presenting with abdominal distension, pain, and significant weight loss over four months. Abdominal CT showed a peritoneal pseudocyst, initially misdiagnosed due to its resemblance to more common abdominal pathologies. The diagnosis of tuberculous peritonitis was confirmed through histopathological analysis. Additionally, a systematic literature review was conducted to identify and analyse similar cases, focusing on clinical presentations, diagnostic methods, and patient outcomes. Our patient exhibited classic symptoms of abdominal TB but was unique due to the absence of prior ventriculoperitoneal shunting, a common factor in similar cases. Our literature review found that such presentations typically result in diagnostic delays averaging five months, complicating patient management and outcomes. This review also underscores the importance of considering tuberculosis in the differential diagnosis of peritoneal pseudocysts, particularly in TB-endemic regions., Conclusion: This case and review emphasize the need for high clinical suspicion and prompt investigation of tuberculosis in patients presenting with atypical abdominal symptoms and pseudocysts. Improved diagnostic strategies, including early use of imaging and pathological evaluations, are essential for timely diagnosis and management, thereby improving patient outcomes in suspected cases of extrapulmonary tuberculosis., (© 2024. The Author(s).)

Aims: Foregut cystic malformations are rare developmental abnormalities, which may involve the hepatopancreaticobiliary tract (HPBT). These cysts are composed of inner ciliated epithelium; subepithelial connective tissue layer; smooth muscle layer; and an outer fibrous layer. While radiopathologic findings are often diagnostic, atypical location and histologic features can pose a diagnostic challenge. We aimed to study ciliated foregut cysts (CFCs) in the HPBT, assess their clinicopathological features with a focus on atypical features., Methods: We collected cases of CFCs involving the HPBT from three large academic medical centres. H&E-stained slides and immunohistochemical stains (where available) were reviewed for each case. Relevant demographic, clinical and pathological information was collected from the medical records., Results: 21 cases were identified. The median age was 53 years (range, 3-78 years). 17 cysts were identified within the liver (segment 4 was the most common location, n=10) and 4 in the pancreas. Cysts were mostly identified incidentally (n=13), abdominal pain was a common symptom (n=5). Cyst size ranged from 0.7 to 17.0 cm (median, 2.5 cm). Radiological findings were available in 17 cases. Cilia were identified in all cases. 19 of 21 cases demonstrated the presence of a smooth muscle layer (thickness, <0.1 mm to 3.0 mm). Three cases showed gastric metaplasia, while one case revealed additional low-grade dysplasia, with features similar to intraductal papillary neoplasm of the bile duct., Conclusions: We highlight clinicopathological features of CFCs in the HPBT. The histomorphology is usually straightforward; however, unusual location and atypical features can pose a diagnostic challenge., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

Background: Splenic cysts are uncommon and very rarely malignant therefore their treatment isn't standardized. In case of symptomatic cysts different surgical approaches have been suggested. Primary malignant lymphoma of the spleen comprises less than 1% of non-Hodgkin's lymphomas. To our knowledge, only 203 cases of splenic large B-cell lymphoma (LBCL) have been reported to date and only 2 of them were fibrin-associated splenic cysts., Case Presentation: 27-year-old model with a 19 × 13 cm splenic cyst without data of malignancy in the preliminary study and therefore treated with laparoscopic deroofing. After histological diagnosis of LBCL with a fibrin/EBV-associated splenic pseudocyst, the patient received 4 cycles of Rituximab and a laparoscopic splenectomy was performed due to resurgence of the pseudocyst. No evidence of malignancy has been found during follow up (EBV viral load every 3 months during the first year, PET-CT every 6 months during the first year and annual afterwards) performed after the splenectomy., Discussion and Conclusions: The value of tumor markers and radiology for diagnosis of splenic cysts is put into question. Only 60 cases of Fibrin-associated LBCL (FA-LBCL) have been described in the literature therefore there are no treatment guidelines for them even though surgery together with systemic treatment has been the prevalent route with good results in most cases., (© 2024. The Author(s).)

Peritoneal inclusion cysts (PICs) are unilocular or multilocular cystic lesions lined by bland mesothelial cells. While most are small and localized, rare examples may be large or multifocal with diffuse peritoneal involvement, causing clinical and even pathologic concern for malignancy. We examined 20 PIC, including 8 large solitary and 12 multifocal lesions. Solitary PIC were found in 7 female and 1 male patients ranging from 19 to 55 (median: 37) years. Expanded collagenous (n=2) or edematous (n=1) areas were occasionally seen in the septae, and 1 had microscopic foci of myxoid stroma. Four had hobnail cells, and 1 had minor areas of papillary mesothelial hyperplasia. Multifocal PICs occurred in 9 female and 3 male patients ranging from 26 to 80 (median: 53) years. Three showed extensive associated fibrosis with entrapment of preexisting adipose tissue, 2 had areas resembling granulation tissue, and 3 had scattered foci of myxoid stroma. Hobnail cells were present in 9, papillary mesothelial hyperplasia in 2, entrapped single cells in 1, and 2 had areas resembling adenomatoid tumors. Two of the multifocal PICs had limited local recurrences at 18 and 21 months. No patients died of disease. Clonal alterations were not identified in any of the tested PICs (mutational and fusion analysis in 5, chromosomal microarray in 1). Despite limited local recurrences, we demonstrate that even large and multifocal PICs may lack identifiable genomic alterations and are associated with benign outcomes., Competing Interests: Conflicts of Interest and Source of Funding: The authors have disclosed that they have no significant relationships with, or financial interest in, any commercial companies pertaining to this article., (Copyright © 2024 Wolters Kluwer Health, Inc. All rights reserved.)

Competing Interests: Declaration of competing interest The authors have no conflicts of interest to declare.

Rationale & Objective: In this pilot study, we hypothesized that autosomal dominant polycystic kidney disease (ADPKD) is characterized by impaired kidney oxidative metabolism that associates with kidney size and cyst burden., Study Design: Cross-sectional study., Setting & Participants: Twenty adults with ADPKD (age, 31±6 years; 65% women; body mass index [BMI], 26.8 [22.7-30.4] kg/m 2 ; estimated glomerular filtration rate [eGFR, 2021 CKD-EPI creatinine], 103±18mL/min/1.73m 2 ; height-adjusted total kidney volume [HTKV], 731±370mL/m; Mayo classifications 1B [5%], 1C [42%], 1D [21%], and 1E [32%]) and 11 controls in normal weight category (NWC) (age, 25±3 years; 45% women; BMI, 22.5 [21.7-24.2] kg/m2; eGFR, 113±15mL/min/1.73m 2 ; HTKV, 159±31mL/m) at the University of Colorado Anschutz Medical Campus., Predictors: ADPKD status (yes/no) and severity (Mayo classifications)., Outcome: HTKV and cyst burden by magnetic resonance imaging, kidney oxidative metabolism, and perfusion by 11 C-acetate positron emission tomography/computed tomography, insulin sensitivity by hyperinsulinemic-euglycemic clamps (presented as ratio of M-value of steady state insulin concentration [M/I])., Analytical Approach: For categorical variables, χ 2 /Fisher's exact tests, and for continuous variables t tests/Mann-Whitney U tests. Pearson correlation was used to estimate the relationships between variables., Results: Compared with NWC individuals, the participants with ADPKD exhibited lower mean±SD M/I ratio (0.586±0.205 vs 0.424±0.171 [mg/kg lean/min]/(μIU/mL), P=0.04), lower median cortical perfusion (1.93 [IQR, 1.80-2.09] vs 0.68 [IQR, 0.47-1.04] mL/min/g, P<0.001) and lower median total kidney oxidative metabolism (0.17 [IQR, 0.16-0.19] vs. 0.14 [IQR, 0.12-0.15] min -1 , P=0.001) in voxel-wise models excluding cysts. HTKV correlated inversely with cortical perfusion (r: -0.83, P < 0.001), total kidney oxidative metabolism (r: -0.61, P<0.001) and M/I (r: -0.41, P = 0.03)., Limitations: Small sample size and cross-sectional design., Conclusions: Adults with ADPKD and preserved kidney function exhibited impaired renal perfusion and kidney oxidative metabolism across a wide range of cysts and kidney enlargements., Funding: Grants from government (National Institutes of Health, Centers for Disease Control and Prevention) and not-for-profit (JDRF) entities., Trial Registration: Registered at ClinicalTrials.gov with study numbers NCT04407481 and NCT04074668., Plain-Language Summary: In our study, we explored how a common genetic kidney condition, autosomal dominant polycystic kidney disease (ADPKD), relates to kidney metabolism. ADPKD leads to the growth of numerous cysts in the kidneys, which can impact their ability to work properly. We wanted to understand the kidneys' ability to process oxygen and blood flow in ADPKD. Our approach involved using advanced imaging techniques to observe kidney metabolism and blood flow in people with ADPKD compared with healthy individuals. We discovered that those with ADPKD had significant changes in kidney oxygen metabolism even when their kidney function was still normal. These findings are crucial as they provide deeper insights into ADPKD, potentially guiding future treatments to target these changes., (Copyright © 2024 National Kidney Foundation, Inc. Published by Elsevier Inc. All rights reserved.)

It is considered that the etiology of endometriosis is retrograde menstruation of endometrial tissue. Although shed endometrial cells are constantly exposed to a challenging environment with iron overload, oxidative stress and hypoxia, a few cells are able to survive and continue to proliferate and invade. Ferroptosis, an iron‑dependent form of non‑apoptotic cell death, is known to play a major role in the development and course of endometriosis. However, few papers have concentrated on the dynamic interaction between autophagy and ferroptosis throughout the progression of diseases. The present review summarized the current understanding of the mechanisms underlying autophagy and ferroptosis in endometriosis and discuss their role in disease development and progression. For the present narrative review electronic databases including PubMed and Google Scholar were searched for literature published up to the October 31, 2023. Autophagy and ferroptosis may be activated at early stages in endometriosis development. On the other hand, excessive activation of intrinsic pathways (e.g., estrogen and mechanistic target of rapamycin) may promote disease progression through autophagy inhibition. Furthermore, suppression of ferroptosis may cause further progression of endometriotic lesions. In conclusion, the autophagy and ferroptosis pathways may play a dual role in disease initiation and progression. The present review discussed the temporal transition of non‑apoptotic cell death regulation during disease progression from retrograde endometrium to early lesions to established lesions.

Purpose: Epididymal cyst lesions (ECLs) include both spermatoceles and epididymal cysts and are often incidentally found on physical exam or scrotal US (SUS). We aimed to determine the association of ECLs and semen parameters among men presenting for fertility evaluation., Materials and Methods: We reviewed men at our institution who had at least 1 semen analysis and SUS available for review between 2002 and 2022. SUS data included testicular measurements, presence or absence of subclinical varicocele, and size and laterality of ECL, if present. Demographic and clinical information including serum testosterone and follicle-stimulating hormone and semen parameters were compared between men with and without ECLs., Results: Among 861 men, 164 (19%) had unilateral right ECL (median 4 mm, interquartile range 3-8 mm), 189 (22%) had unilateral left ECL (median 4 mm, interquartile range 3-9 mm), and 113 (13%) had bilateral ECL. Patients with ECLs were significantly older than men without ECLs at the time of evaluation but had no statistically significant difference in semen volume, sperm concentration, sperm motility, sperm morphology, total motile sperm count, or serum hormonal values. Analysis of men with unilateral and bilateral ECLs showed that ECL size and laterality did not significantly correlate with any semen parameter evaluated., Conclusions: We found no association between ECLs and semen parameters. Patients should be counseled toward conservative management with observation for asymptomatic ECLs in the setting of fertility evaluation.

Background: We conducted this study to clarify the magnetic resonance imaging (MRI) characteristics of lobular endocervical glandular hyperplasia (LEGH) and Nabothian cysts., Methods: This study included 48 patients who underwent hysterectomy at our institution between 2016 and 2020 for suspected LEGH. Histopathological studies confirmed the presence of 25 Nabothian cysts and 23 cases of LEGH. We retrospectively analyzed five characteristic MRI findings: (1) located at the upper cervical canal, (2) positioned within the cervical stroma, (3) not circumscribing the cervical canal, (4) low- to iso-intensity on T1-weighted images (T1WI), and (5) "cosmos" or "microcystic" pattern. We compared the diagnostic accuracy of these findings for LEGH and Nabothian cysts using sensitivity, specificity, and predictive values. Combinations of findings were also calculated., Results: The characteristics "cosmos" or "microcystic" pattern, lesion not circumscribing the cervical canal, and low/iso-intensity on T1WI had a sensitivity and specificity greater than 50%. The sensitivity was 73.9% and specificity 84.0% when a combination of "cosmos" or "microcystic" pattern and lesion not circumscribing the cervical canal was present., Conclusion: The coexistence of a "cosmos" or "microcystic" pattern and not circumscribing the cervical canal was the most characteristic finding that distinguished LEGH from Nabothian cysts. When neither of these findings is present, Nabothian cyst can be suspected., (© 2024. The Author(s).)

Rationale: Multilocular cystic hemangioma is a rare benign tumor classified as an atypical hemangioma. Currently, there are limited imaging reports available, and the imaging characteristics can be challenging to distinguish from other malignant multilocular cystic liver diseases such as cystadenocarcinoma, necessitating confirmation through pathological diagnosis. Here, we discuss the imaging features of 3 cases of multilocular cystic hemangiomas., Patient Concerns and Diagnoses: Case 1 was a 24-year-old young female, and Case 2 involved a 60-year-old elderly male. Both patients were asymptomatic and physical examination revealed hepatic space-occupying lesions. Imaging findings revealed multilocular cystic lesions in the left liver with septa, calcification, a high diffusion-weighted magnetic resonance imaging (DWI) signal at the edge of the lesion, and progressive enhancement of the cyst wall and septa. Case 3 involved a 50-year-old male patient with epigastric distending pain for 1 month and sudden severe abdominal pain for 14 hours. Imaging results revealed a multilocular cystic lesion in the left liver with septa and tumor bleeding, a high DWI signal, and an enhanced cyst wall and septa. The pathological diagnosis confirmed a hepatic hemangioma., Interventions: All 3 patients underwent liver tumor resection., Outcomes: All 3 patients recovered successfully without any intraoperative or postoperative complications during the follow-up periods of 5 years, 6 months, and 5 months. There were no signs of recurrence., Lessons: Liver imaging revealed multilocular cystic lesions with features, such as compartmentalization, calcification, or bleeding. Multilocular cystic hemangiomas should be considered in imaging diagnosis. Enhancing our understanding of multilocular cystic hemangiomas can aid in improving the differential diagnosis of other malignant multilocular cystic liver diseases, ultimately reducing unnecessary liver resection., Competing Interests: The authors have no conflicts of interest to disclose., (Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.)

Objectives: The aim of this study was to investigate whether the presence and size of fibrous cysts affected postoperative results in patients undergoing hip arthroscopy., Patients and Methods: Between January 2010 and December 2019, a total of 261 patients (138 males, 123 females; mean age: 39.5±11.9 years; range, 18 to 66 years) who underwent hip arthroscopy with the diagnosis of cam-pincer-mixed-type femoroacetabular impingement (FAI) and labral pathologies were retrospectively analyzed. The study groups (impingements and labral pathologies) and the presence of cyst (or cyst size: <5 mm, 5-8 mm, >8 mm) were used as the fixed effects, and the analysis was adjusted for baseline age, sex, and preoperative scores. Pre- and postoperative modified Harris Hip Score (mHHS) and Visual Analog Scale (VAS) scores that were applied to all patients were used as an indication of clinical results., Results: The mean preoperative mHHS score of the patients with a cyst was significantly lower compared to the patients without a cyst (56.8±12.3 vs. 60.3±12.7, p=0.026). The mean change in the mHHS score and the mean percentage change in VAS score were significantly higher in the patients with a cyst compared to the patients without a cyst (mHHS score: 28.1±14.0 vs. 22.5±14.1, p=0.002; VAS score: 61.9±30.2 vs. 52.6±47.4, p=0.038). The increase in mHHS score over time for patients with a cyst was significantly higher than the patients without cysts in the pincer group (38.1±11.1 vs. 19.3±13.5, p<0.001). The patients with a cyst size of >8 mm had a significantly higher increase in the mHHS scores compared to the patients with a cyst size of <5 mm (29.5±12.9 vs. 23.5±13.8, p=0.043)., Conclusion: Subchondral cysts in the femoral head and neck junction accompanied cam-type and mixed-type FAI, while subchondral cysts in the acetabulum accompanied pincer-type impingement. In all groups, the mean increase in mHHS scores and the mean decrease in VAS scores were higher in patients with subchondral cysts than in patients without cysts. In patients with subchondral cysts, if the lesion causing FAI is treated arthroscopically, it can positively affect the functional results.

Background: Our aim was to determine whether the Apparent Diffusion Coefficient is able to predict the presence of a symptomatic pineal cyst by detecting cerebral edema., Methods: We retrospectively analyzed MRIs of 45 patients with pineal cysts before and after resection and 51 patients without pineal cysts, comparing ADC values of thalamus, central, periventricular and subcortical white matter. Furthermore we evaluated cyst size and morphology and analyzed its correlation to ADC values in corresponding patients., Results: Differences between patients with symptomatic pineal cyst and control group were not significant (p = 0.200 - 0.968). ADC ratios did not change significantly after resection of the cyst (p = 0.575 - 0.862). Cyst size showed no significant correlation to ADC ratios (p = 0.071 - 0.918). Raw data analyses revealed more significance, especially periventricularly and in central white matter, which resulted in significant interhemispheric differences in ADC ratios in both subgroups (p < 0.001 and p = 0.031). MRI of 1.5T showed consistently higher values than 3T but mostly insignificant., Conclusion: Our analysis revealed no evidence that pineal cysts lead to intracerebral edema caused by venous compression. Since variability was higher than the differences seen, ADC sequences do not appear to be an appropriate diagnostic tool for symptomatic pineal cysts., (© 2024. The Author(s).)

Colitis cystica profunda (CCP) is a rare, uncommon and nonneoplastic condition that can occur anywhere in gastrointestinal tract, but its main occurrence is in the rectum and sigmoid colon. It is characterized by the presence of mucin filled cysts, lined by benign epithelium, beneath the muscularis mucosae, usually confined to the submucosa, and it can clinically and radiologically mimic a neoplasm. Here we report a rare case of CCP in a patient with a 2-months history of abdominal pain and severe anemia, associated with diverticulosis. The knowledge of this entity and its differential diagnosis, in particular with the intestinal mucinous adenocarcinoma, is necessary, as it can be a clinically and histological mimic of a malignant neoplasm., (Copyright © 2024 Società Italiana di Anatomia Patologica e Citopatologia Diagnostica, Divisione Italiana della International Academy of Pathology.)

A new gene-cell construct expressing nerve growth factor (NGF) has been developed. After obtaining engineered adenovectors Ad5-RGD-CAG-NGF and Ad5-RGD-CAG-EGFP, transduction efficiency and transgene expression were studied and multiplicity of infection was determined. The efficacy of transduced human olfactory ensheathing cells expressing NGF in restoring motor activity in rats has been shown in a limited period of time. Improved rat hindlimb mobility and cyst size reduction after gene-cell construct transplantation were more likely due to the cellular component of the construct., (© 2024. Springer Science+Business Media, LLC, part of Springer Nature.)

"Cysts of the ligamentum flavum (cysts-LF)" is the term for non-neoplastic cystic lesion involving LF. The aim of the present study was to elucidate the histopathological characteristics and pathogenesis of "cysts-LF". Herein, we defined cysts-LF as spinal cysts containing degenerative LF components. From archival cases, we investigated 18 symptomatic cysts-LF surgically removed from 18 patients (13 males and five females; median age 68.5 years [range, 42-86 years]). The elastic fibers of LF components in the wall were separated and/or torn, and cyst walls were accompanied by chondroid metaplasia (17 cases), myxoid changes (13 cases), ossification (11 cases), amyloid deposits (14 cases), hemosiderosis (six cases), granular/smudgy calcification (four cases), synovial cell linings (three cases), and severe inflammatory infiltrates (one case). These histologic features of our cysts-LF were shared by previously reported "cysts-LF." Fourteen cysts-LF demonstrated vascular stenosis/occlusion, and eight showed thick hyalinized vessels, suggesting local circulatory insufficiency. Eight cases (44%) exhibited lipomembranous fat necrosis, accompanied by hyalinized vascular changes (p = 0.003). Ischemic conditions were observed in nearly half of the present cysts-LF, and may be one of the main contributing factors for the formation of cysts-LF, via degeneration and cystic changes in the LF., (© 2024 Japanese Society of Pathology and John Wiley & Sons Australia, Ltd.)

Background: Cystic and nodular lung diseases encompass a broad spectrum of diseases with different etiologies and clinicoradiological presentations. Their differentiation is crucial for patient management but can be complex due to diseases with features of both categories and overlapping radiological patterns., Objective: This study aims to describe the imaging features of cystic and nodular lung diseases in high-resolution computed tomography (CT) in detail-primarily based on their etiology-in order to allow a more accurate differential diagnosis of these diseases., Materials and Methods: A narrative review based on current literature on the topic was conducted from a clinicoradiological perspective., Results: This paper systematically categorizes the differential diagnosis of cystic and nodular lung disease and provides insights into their radiological patterns and etiologies. It highlights the role of CT in the diagnosis of these diseases and emphasizes the importance of multidisciplinary panels combining expertise from radiology, pulmonology, rheumatology, and pathology., Conclusion: Reliable differential diagnosis of cystic and nodular lung diseases, particularly based on their radiological features alone, remains difficult due to their overlapping and dynamic nature. Multidisciplinary boards should be the clinical standard for accurate work-up of these diseases, as they combine the medical history, symptoms, radiological findings, and, if necessary, histopathological examinations, thus providing a more robust framework for diagnosis and management., (© 2024. The Author(s), under exclusive licence to Springer Medizin Verlag GmbH, ein Teil von Springer Nature.)

Background : Inflammatory lesions and cysts are the most common Bartholin glands pathologic diagnoses. There have been very few reports of benign solid lesions of Bartholin glands such as nodular hyperplasia. Materials and methods : Thirty-two lesions diagnosed as Bartholin duct cyst in the gynecologic specialized institute were reviewed by two expert gynecological pathologists. Results : Using specific criteria, 7 qualified as nodular hyperplasia, 25 as Bartholin duct cyst. The average age of the patients with nodular hyperplasia was 31 years (range, 24 to 43). These lesions were solid, solid and cystic or purely cystic, had a mean maximal dimension of 3.3 cm, and were frequently thought to be Bartholin cysts on clinical examination. Four lesions were diagnosed as Bartholin duct cysts, while in the review they were considered as nodular hyperplasia. Conclusion : Nodular hyperplasia of Bartholin gland may be more common than is reported in the literature. This study showed that the diagnosis of nodular hyperplasia has been overlooked due to limited literature sources., Competing Interests: Declaration of Conflicting InterestsThe authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

A group of genetic diseases known as polycystic liver disease (PLD) are distinguished by the gradual development of fluid-filled hepatic cysts formed from cholangiocytes and commonly related to primary cilia defects. The NAD salvage pathway, which sustains cellular bioenergetics and supplies a required substrate for tasks important to rapidly multiplying cells, has a rate-limiting phase that is mediated by nicotinamide phosphoribosyltransferase (NAMPT). In this study, the efficacy and mechanisms of action of FK866, a novel, high-potency NAMPT inhibitor with a good toxicity profile, were assessed. NAMPT-siRNA and FK866 reduced NAD levels and inhibited the proliferation of PLD cells in a dose-dependent manner. Notably, this pharmacologic and siRNA-mediated suppression of NAMPT was less effective in normal cells at the same concentrations. The addition of nicotinamide mononucleotide (NMN), a byproduct of NAMPT that restores NAD concentration, rescued the cellular viability of PLD cells and verified the on-target action of FK866. In FK866-treated PLD cells, mitochondrial respiration and ATP production were impaired and reactive oxygen species production was induced. Importantly, FK866 treatment was associated with improved effects of octreotide, a drug used for PLD treatment. As a result, the use of NAMPT inhibitors, including FK866 therapy, offers the possibility of a further targeted strategy for the therapeutic treatment of PLD., Competing Interests: Disclosure Statement None declared., (Copyright © 2024 American Society for Investigative Pathology. Published by Elsevier Inc. All rights reserved.)

Background: Adrenal cysts are rare and appropriate management is unclear due to a lack of data on their natural history. Understanding adrenal cyst growth patterns would assist in clinical management., Methods: This single-institution study included all adult patients diagnosed with simple adrenal cysts between 2004 and 2021. Baseline characteristics and outcomes of those who underwent resection (ADX) or observation (OBS) were compared using the chi-squared test, student's t-test, and Wilcoxon rank-sum test. Growth curves and sensitivity analysis were plotted for all patients who had follow-up imaging., Results: We identified 77 patients with imaging-confirmed adrenal cysts. The majority were female (75.3%) and more than half were white (55.8%). One-third of patients underwent ADX, and the remaining were observed. ADX patients were younger (median age [IQR]: 55.5 y [45.0-68.2 y] vs. 44.2 y [38.7-55.0 y], p = 0.01) and more likely to be Hispanic (12% vs. 0%, p = 0.05). ADX patients presented with larger cysts (5.6 vs. 2.6 cm, p = 0.002). The median time from diagnosis to last follow-up was 1.1 y for ADX and 4.1 y for OBS. Average growth for OBS was 0.3 cm/y, while average growth for ADX was 3.9 cm/y. In ADX patients, cysts >10 cm grew significantly faster than cysts <10 cm (median growth rate 13.2 cm/y vs. 0.3 cm/y, p < 0.05). There was no adrenal malignancy diagnosis, hyperfunctionality, or observation-related complications (e.g., rupture)., Conclusion: While size >4-6 cm has guided surgical referral for solid adrenal masses, this study demonstrates a size threshold of 10 cm, below which asymptomatic, simple adrenal cysts can safely be observed., (© 2024 International Society of Surgery/Société Internationale de Chirurgie (ISS/SIC).)

Pineal cysts are frequently encountered as incidental findings in magnetic resonance imaging, usually devoid of symptoms, yet some patients exhibit symptomatic manifestations possibly associated with the cyst, even in the absence of hydrocephalus. The etiology of these symptoms remains contentious. This study aims to investigate the presence of lymphatic endothelial cell (LEC) markers and indications of inflammation or immune response within the pineal cysts of patients experiencing symptomatic non-hydrocephalic presentations. Eight patients who underwent surgical excision of their cysts were included in the study. Immunohistochemistry was utilized to assess the expression of LYVE-1, PDPN, and VEGFR3 as LEC markers, alongside IL-6 and CD3 for indications of inflammation or immune activity. Our analysis revealed an absence of inflammatory markers or immune response. However, a distinct expression of VEGFR3 was observed, likely localized to neurons within the pineal cyst tissue. We propose that these VEGFR3+ neurons within the pineal cyst may contribute to the headache symptoms reported by these patients. Further investigations are warranted to substantiate this hypothesis., Competing Interests: Declaration of competing interest The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper., (Copyright © 2024 The Authors. Published by Elsevier B.V. All rights reserved.)

Background: Autosomal dominant polycystic kidney disease (ADPKD) is the most prevalent genetic kidney disorder. While metformin has demonstrated the ability to inhibit cyst growth in animal models of ADPKD via activation of adenosine monophosphate-activated protein kinase (AMPK), its effectiveness in humans is limited due to its low potency. This study explored the impact of HL156A, a new and more potent AMPK activator, in a mouse model of ADPKD., Methods: To investigate whether HL156A inhibits the proliferation of renal cyst cells in ADPKD in vitro, exogenous human telomerase reverse transcriptase (hTERT)-immortalized renal cyst cells from ADPKD patients were treated with HL156A, and an MTT (dimethylthiazol-diphenyltetrazolium bromide) assay was performed. To assess the cyst-inhibitory effect of HL156A in vivo, we generated Pkd1 conditional knockout (KO) mice with aquaporin 2 (AQP2)-Cre, which selectively expresses Cre recombinase in the collecting duct. The effectiveness of HL156A in inhibiting cyst growth and improving renal function was confirmed by measuring the number of cysts and blood urea nitrogen (BUN) levels in the collecting duct-specific Pkd1 KO mice., Results: When cyst cells were treated with up to 20 µM of metformin or HL156A, HL156A reduced cell viability by 25% starting at a concentration of 5 µM, whereas metformin showed no effect. When AQP2-Cre male mice were crossed with Pkd1 flox/flox female mice, and when AQP2-Cre female mice were crossed with Pkd1 flox/flox male mice, the number of litters produced by both groups was comparable. In collecting duct-specific Pkd1 KO mice, HL156A was found to inhibit cyst growth, reducing both the number and size of cysts. Furthermore, it was confirmed that kidney function improved as HL156A treatment led to a reduction in elevated BUN levels. Lastly, it was observed that the increase in AMPK phosphorylation induced by HL156A decreased ERK phosphorylation and α-SMA expression., Conclusion: HL156A has potential as a drug that can restore kidney function in ADPKD patients by inhibiting cyst growth.

Competing Interests: Conflict of interest All authors have no conflict of interest to disclose.

Cystic degeneration of the fibrous dysplasia is a very rare clinical condition and may present with loss of vision when it involved the skull base. A 12-year-old female child presented with an enlargement of the skull. She was diagnosed as large skull base and skull vault tumor. She underwent partial removal of the tumor, and custom-made titanium implant was inserted. The diagnosis was fibrous dysplasia. Two years after the initial diagnosis, she presented with total loss of vision at her right eye. Radiological imaging confirmed the cystic degeneration within the tumor. She re-operated and the cyst fluid was evacuated in association with the removal of cyst wall. The diagnosis was the cystic degeneration of the fibrous dysplasia. Her vision was improved a few days after the surgery. Fibrous dysplasia of the skull base should be closely followed-up in order to prevent severe visual complications., (© 2024. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

BACKGROUND. Concern may exist that pulmonary lesions associated with cystic airspaces are at risk of increased biopsy complications or lower biopsy accuracy given challenges in targeting tissue abutting or intermingled with the cystic airspaces. OBJECTIVE. The purpose of this study was to evaluate the safety and diagnostic performance of CT-guided core needle biopsy (CNB) of pulmonary lesions associated with cystic airspaces. METHODS. This retrospective study included 90 patients (median age, 69.5 years; 28 women, 62 men) who underwent CT-guided CNB of pulmonary lesions associated with cystic airspaces (based on review of procedural images) from February 2010 to December 2022 and a matched control group (2:1 ratio) of 180 patients (median age, 68.0 years; 56 women, 124 men) who underwent CNB of noncystic noncavitary lesions during the same period. The groups were compared in terms of complications, nondiagnostic biopsies (i.e., nonspecific benignities, atypical cells, or insufficient specimens), and CNB diagnostic performance for detecting malignancy using as reference the final diagnosis from a joint review of all available records. For lesions associated with cystic airspaces that underwent surgical resection after CNB, histologic slides were reviewed to explore the nature of the cystic airspace. RESULTS. The final diagnosis was malignant in 90% (81/90) of lesions associated with cystic airspaces and 92% (165/180) of noncystic noncavitary lesions. Patients with lesions associated with cystic airspaces and patients with noncystic noncavitary lesions showed no significant difference in frequency of complications (overall: 40% [36/90] vs 38% [68/180], p = .79; major: 4% [4/90] vs 6% [10/180], p = .78; minor: 36% [32/90] vs 32% [58/180], p = .59), frequency of nondiagnostic biopsies (12% [11/90] vs 9% [16/180], p = .40), or diagnostic performance (accuracy: 94% [85/90] vs 97% [175/180], p = .50; sensitivity: 94% [76/81] vs 97% [160/165], p = .50; specificity: 100% [9/9] vs 100% [15/15]; p > .99), respectively. All false-negative results for malignancy in both groups occurred in patients with nondiagnostic CNB results. Among lesions associated with cystic airspaces that were resected after CNB (all malignant), the cystic airspaces most commonly represented tumor degeneration (22/31 [71%]). CONCLUSION. CT-guided CNB is safe and accurate for assessing pulmonary lesions associated with cystic airspaces. CLINICAL IMPACT. CNB may help avoid a missed or delayed cancer diagnosis in pulmonary lesions with cystic airspaces.

Focal liver lesions (FLLs) have become an increasingly common finding on abdominal imaging, especially asymptomatic and incidental liver lesions. Gastroenterologists and hepatologists often see these patients in consultation and make recommendations for management of multiple types of liver lesions, including hepatocellular adenoma, focal nodular hyperplasia, hemangioma, and hepatic cystic lesions including polycystic liver disease. Malignancy is important to consider in the differential diagnosis of FLLs, and healthcare providers must be familiar with the diagnosis and management of FLLs. This American College of Gastroenterology practice guideline uses the best evidence available to make diagnosis and management recommendations for the most common FLLs., (Copyright © 2024 by The American College of Gastroenterology.)

The gradually progressive solitary cystic-solid mass of chest CT scans is highly suggestive of lung cancer. We report a case of a 29-year-old woman with a persistent cystic-solid lesion in the right upper lobe. A chest CT scan showed a 35 mm × 44 mm × 51 mm focal cystic-solid mass in the anterior segment of the right upper lobe. The size of lesion had increased over 3 years, especially for the solid component. The right upper lobe pneumonectomy was performed. Postoperative pathological examination showed placental transmogrification of the lung, which is a rare cause of pulmonary cystic lesion., (© 2024 The Author(s). The Clinical Respiratory Journal published by John Wiley & Sons Ltd.)

Background: Pleuropulmonary blastoma (PPB), the hallmark tumour associated with DICER1 -related tumour predisposition, is characterised by an age-related progression from a cystic lesion (type I) to a high-grade sarcoma with mixed cystic and solid features (type II) or purely solid lesion (type III). Not all cystic PPBs progress; type Ir (regressed), hypothesised to represent regressed or non-progressed type I PPB, is an air-filled, cystic lesion lacking a primitive sarcomatous component. This study aims to evaluate the prevalence of non-progressed lung cysts detected by CT scan in adolescents and adults with germline DICER1 pathogenic/likely pathogenic (P/LP) variants., Methods: Individuals were enrolled in the National Cancer Institute Natural History of DICER1 Syndrome study, the International PPB/ DICER1 Registry and/or the International Ovarian and Testicular Stromal Tumor Registry. Individuals with a germline DICER1 P/LP variant with first chest CT at 12 years of age or older were selected for this analysis., Results: In the combined databases, 110 individuals with a germline DICER1 P/LP variant who underwent first chest CT at or after the age of 12 were identified. Cystic lung lesions were identified in 38% (42/110) with a total of 72 cystic lesions detected. No demographic differences were noted between those with lung cysts and those without lung cysts. Five cysts were resected with four centrally reviewed as type Ir PPB., Conclusion: Lung cysts are common in adolescents and adults with germline DICER1 variation. Further study is needed to understand the mechanism of non-progression or regression of lung cysts in childhood to guide judicious intervention., Competing Interests: Competing interests: Dr Hill is the owner of ResourcePath LLC, a company which does research and development of laboratory tests including for DICER1-related cancers. The work is unrelated to the information presented in this article. Dr Stewart provides telegenetics services for Genome Medical, Inc, in accordance with relevant National Cancer Institute policies. The remaining authors have no conflicts to disclose., (© Author(s) (or their employer(s)) 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

Abnormal lung development can cause congenital pulmonary cysts, the mechanisms of which remain largely unknown. Although the cystic lesions are believed to result directly from disrupted airway epithelial cell growth, the extent to which developmental defects in lung mesenchymal cells contribute to abnormal airway epithelial cell growth and subsequent cystic lesions has not been thoroughly examined. In the present study using genetic mouse models, we dissected the roles of bone morphogenetic protein (BMP) receptor 1a (Bmpr1a)-mediated BMP signaling in lung mesenchyme during prenatal lung development and discovered that abrogation of mesenchymal Bmpr1a disrupted normal lung branching morphogenesis, leading to the formation of prenatal pulmonary cystic lesions. Severe deficiency of airway smooth muscle cells and subepithelial elastin fibers were found in the cystic airways of the mesenchymal Bmpr1a knockout lungs. In addition, ectopic mesenchymal expression of BMP ligands and airway epithelial perturbation of the Sox2-Sox9 proximal-distal axis were detected in the mesenchymal Bmpr1a knockout lungs. However, deletion of Smad1/5, two major BMP signaling downstream effectors, from the lung mesenchyme did not phenocopy the cystic abnormalities observed in the mesenchymal Bmpr1a knockout lungs, suggesting that a Smad-independent mechanism contributes to prenatal pulmonary cystic lesions. These findings reveal for the first time the role of mesenchymal BMP signaling in lung development and a potential pathogenic mechanism underlying congenital pulmonary cysts., Competing Interests: YL, KC, JC, HC, HW, MT, BG, MK, MP, YM, WS No competing interests declared, (© 2023, Luo et al.)

Objective: The neck region is a common site for solitary cystic neck mass (SCNM) of various etiologies, including congenital, inflammatory, and neoplastic. In adults, the primary focus is excluding malignancy. The objective of this study was to retrospectively analyze the accuracy of available diagnostic technologies for the differentiation of benign and malignant SCNM in adult patients. The study aimed to develop new clinical practice guidelines for evaluating and managing SCNM., Methods: The primary predictive variables were the diagnostic utilities of fine-needle aspiration cytology (FNAC), ultrasound (U/S), multislice computed tomography, and magnetic resonance imaging. The study's endpoint was the overall diagnostic accuracy in differentiating between benign and malignant SCNM. The final diagnosis was based on histopathology., Results: The study included 79 adult patients: 55 (69.62%) male and 24 (30.38%) female ( P <0.05). The mean age at presentation was 42.1 years (range: 18-84 years). Solitary cystic neck mass was distributed in the anterior neck region in 30 (37.97%) patients and the posterolateral neck regions in 49 (62.03%) patients ( P <0.05). The posterolateral neck regions had a significantly higher rate of malignant SCNM than the anterior neck region [19/49 (38.78%) versus 1/30 (3.33%)] ( P <0.05). There was no statistically significant difference between the U/S+FNAC and U/S+FNAC+multislice computed tomography and/or magnetic resonance imaging groups in differentiating benign and malignant SCNM (40/42 versus 36/37, P >0.05). "Violated neck" was recorded in 2 cases., Conclusion: A systematic investigation protocol should be applied to evaluate adult patients with SCNM., Competing Interests: The authors report no conflicts of interest., (Copyright © 2024 by Mutaz B. Habal, MD.)

Hepatic cystadenoma is a rare disease, accounting for about 5% of all cystic lesions, with a high tendency of malignant transformation. The preoperative diagnosis of cystadenoma is difficult, and some cystadenomas are easily misdiagnosed as hepatic cysts at first. Hepatic cyst is a relatively common liver disease, most of which are benign, but large hepatic cysts can lead to pressure on the bile duct, resulting in abnormal liver function. To better understand the difference between the microenvironment of cystadenomas and hepatic cysts, we performed single-nuclei RNA-sequencing on cystadenoma and hepatic cysts samples. In addition, we performed spatial transcriptome sequencing of hepatic cysts. Based on nucleus RNA-sequencing data, a total of seven major cell types were identified. Here we described the tumor microenvironment of cystadenomas and hepatic cysts, particularly the transcriptome signatures and regulators of immune cells and stromal cells. By inferring copy number variation, it was found that the malignant degree of hepatic stellate cells in cystadenoma was higher. Pseudotime trajectory analysis demonstrated dynamic transformation of hepatocytes in hepatic cysts and cystadenomas. Cystadenomas had higher immune infiltration than hepatic cysts, and T cells had a more complex regulatory mechanism in cystadenomas than hepatic cysts. Immunohistochemistry confirms a cystadenoma-specific T-cell immunoregulatory mechanism. These results provided a single-cell atlas of cystadenomas and hepatic cyst, revealed a more complex microenvironment in cystadenomas than in hepatic cysts, and provided new perspective for the molecular mechanisms of cystadenomas and hepatic cyst., Competing Interests: Declaration of competing interest This paper is a full-length manuscript that has not simultaneously been submitted elsewhere for print or electronic publication. This submission has been agreed by all the co-authors for this paper. All the co-authors declare no conflict of interest., (Copyright © 2024 Elsevier Ltd. All rights reserved.)

Foregut duplication cyst is most commonly seen in the posterior mediastinum without communication with adjacent organs or presence of other malformations and typically shows ectopic gastric or respiratory epithelium. The finding of ectopic pancreatic tissue is extremely rare. A 15-year-old female patient presented with elevated blood pressure, intermittent right flank pain for 18 months. An abdominal MRI revealed a 4 cm right cystic retroperitoneal mass, initially thought to arise from the right adrenal gland. Intraoperatively, the mass was centered in the retroperitoneum between the right adrenal gland and kidney, without attachments to these organs and no communication with other structures, and it was completely excised. Gross examination revealed a 3.5 cm well-circumscribed solid and cystic mass with orange-white cut surface and cloudy fluid surrounded by variable amounts of adipose tissue. On microscopic examination, the cysts were lined by respiratory and gastric antral/oxyntic-type mucosa, surrounded by muscularis mucosae, submucosal tissue, muscularis propria, and perienteric tissue. Within the muscular propria, exocrine pancreatic tissue was also noted. The patient is well with 7-months of follow up. This case is the first one reporting gastric, respiratory, and pancreatic epithelia., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.