Case Series Research Articles

Endometrial carcinoma with cervical stromal invasion: Three case reports

BACKGROUND Endometrial cancer is a kind of well-known tumors of female genitourinary system. Cervical stromal invasion is an adverse factor for poor prognosis of endometrial cancer. There is still controversy regarding the use of magnetic resonance imaging (MRI) in the diagnosis of cervical stromal invasion of endometrial cancer. The diagnosis of cervical stromal invasion varies significantly between different observers and institutions. We present a limited case series of the particular pattern of endometrial cancer, which infiltrates the cervical stroma and is often overlooked. CASE SUMMARY We present three cases of endometrial carcinoma with cervical stromal invasion with cancer-free uterine cavity. One patient, a reproductive-aged woman, exhibited irregular menstruation and was diagnosed with endometrial polyps by hysteroscopy and segmental curettage. A MRI scan revealed polypoid nodules within the internal cervical orifice. The other two cases were postmenopausal women who presented with abnormal vaginal bleeding. Hysteroscopy and segmental curettage suggested atypical hyperplasia of the endometrium. MRI scans did not detect any malignant signs in the endometrium. In one case, a non-thickened endometrium was observed, while in another, hyperplasia of the endometrium was seen. Notably, none of these patients had malignant tumors identified in the uterine cavity via MRI scans. However, postoperative pathological results following hysterectomy consistently indicated cervical stromal invasion. CONCLUSION Cervical stromal invasion is easily missed if no cancer is found in the uterine body on MRI. Immunohistochemistry of endoscopic curettage specimens should be conducted to avoid underestimation of the disease.

Comparison of ocular biometric parameters between two swept-source optical coherence tomography devices and Scheimpflug tomography in patients with cataract

AIM: To assess and compare the variations and agreements across different ocular biometric parameters using swept-source optical coherence tomography (SS-OCT) and Scheimpflug tomography in patients diagnosed with cataract. METHODS: This prospective case series was conducted at Tianjin Medical University Eye Hospital. In total, 212 eyes from 212 patients scheduled for phacoemulsification were included. Eyes were evaluated preoperatively using two SS-OCT devices (IOLMaster700 and CASIA2) and Scheimpflug tomography (Pentacam). Central corneal thickness (CCT), anterior chamber depth (ACD), aqueous depth (AQD), white-to-white distance (WTW), flat simulated keratometry (Kf), steep simulated keratometry (Ks), mean keratometry (Km), and total corneal keratometry (TKm) were measured. Intraclass correlation coefficient (ICC), 95% confidence intervals (CI) and limits of agreement (LoA) widths were conducted to assess differences and correlations between devices. RESULTS: All parameters, except for Ks, were significantly different. Pairwise comparison revealed no significant differences between keratometry obtained by IOLMaster 700 and Pentacam. LoA widths of all paired comparisons for Ks were >0.80 D. Except for WTW between IOLMaster 700 and CASIA2 and between CASIA2 and Pentacam, other Pearson’s coefficients between devices showed a strong correlation (all r>0.95). The ICC of WTW (ICC=0.438, 95%CI 0.167-0.625) showed poor reliability. The reliability of CCT, ACD, and AQD was excellent (all ICC>0.95), whereas that of TKm was good (ICC=0.827, 95%CI 0.221-0.939). A significant linear correlation was also observed among devices. CONCLUSION: The ocular parameters derived from the use of IOLMaster700, CASIA2, and Pentacam exhibit significant discrepancies; as such, measurements from these devices should not be deemed as interchangeable.

Accuracy comparison of total keratometry and standard keratometry in intraocular lens power calculations for post-corneal refractive surgery cataract patients

Objective: To compare the accuracy of intraocular lens (IOL) power calculations using total keratometry (TK) versus standard keratometry (K) in post-corneal refractive surgery cataract patients. Methods: This retrospective case series study included 30 patients (36 eyes) with a history of laser corneal refractive surgery who underwent cataract extraction and IOL implantation at Qingdao Eye Hospital, Affiliated to Shandong First Medical University, from September 2022 to December 2023. The cohort comprised 16 males and 14 females, with an average age of (53.6±8.1) years. IOL power was calculated using the K-based Haigis-L and Barrett True-K formulas, as well as the TK-based Haigis and Barrett Universal Ⅱ formulas. Postoperative objective refraction was performed to obtain the actual refractive status of the operated eyes. The refractive prediction error (RPE) was defined as the difference between the actual spherical equivalent and the predicted refraction. The absolute value of the RPE was taken as the refractive absolute error (RAE). Differences in errors calculated by the four formulas were compared. Results: TK showed good consistency with K, with TK being on average 0.50 D lower than K. Analysis of variance revealed statistically significant differences in RPE among the four formulas (P<0.001). The RPE for the TK-based Haigis formula was (0.17±0.09) D, and for the Barrett Universal Ⅱ formula, it was (0.21±0.11) D, both significantly better than the K-based Haigis-L formula (-0.61±0.12) D and Barrett True-K formula (-0.57±0.11) D (all P<0.001). The percentage of eyes with postoperative RPE<±1.00 D was higher for the TK-based Haigis (92%, 33 eyes) and Barrett Universal Ⅱ (86%, 31 eyes) formulas compared to the TK-based Barrett True-K (75%, 27 eyes) and Haigis-L formulas (67%, 24 eyes), with statistically significant differences (P<0.05). Conclusions: Compared with K, TK improves the accuracy of IOL power calculation in post-corneal refractive surgery patients. Both the TK-based Barrett Universal Ⅱ and Haigis formulas demonstrate high accuracy.

Explore the safety and efficiency of mild myopia retention in patients with myopia and presbyopia

Objective: To explore the corrective effects of a personalized corneal refractive surgery design that retains mild myopia in patients over 40 years old with refractive errors and presbyopia. Methods: A retrospective case series study was conducted, including 60 patients (120 eyes) over 40 years old who underwent corneal refractive surgery at Peking Union Medical College Hospital l from January 2023 to December 2023. The patients were divided into two groups based on their preference: Group A (retained mild myopia) and Group B (fully corrected), with 30 patients (60 eyes) in each group. Preoperative and postoperative visual acuity, subjective refraction, slit-lamp examination, corneal topography, and intraocular pressure were assessed at 1 day, 1 week, 1 month, 3 months, 6 months, and 12 months after surgery. The effectiveness and safety indices were calculated based on visual acuity before and after surgery. The National Eye Institute Refractive Quality of Life questionnaire was used to evaluate patient satisfaction and postoperative visual symptoms. Results: There were no significant differences in preoperative uncorrected distance visual acuity (UDVA), corrected distance visual acuity (CDVA), spherical equivalent (SE), corneal thickness, and intraocular pressure between the two groups (all P>0.05). At the final follow-up, the proportions of eyes with UDVA≥0.8 and≥1.0 were 93.3% (56/60) and 60.0% (36/60) in Group A, and 100% (60/60) and 83.3% (50/60) in Group B, respectively. The SE was significantly different between Group A [(-0.35±0.52) D] and Group B [(-0.07±0.55) D] (P<0.05). Near visual acuity was better in Group A than in Group B (P<0.05). The effectiveness indices were 0.96±0.23 and 0.99±0.12, and the safety indices were 1.02±0.11 and 1.02±0.07 for Groups A and B, respectively. Both groups had high overall satisfaction, but Group A had higher scores for near vision, reading, and computer screen viewing. Conclusion: The personalized corneal refractive surgery design that retains mild myopia provides good corrective effects for patients over 40 years old with refractive errors, improving patient satisfaction and quality of life.

Exploring Uncommon Neurologic Complication in Young Diabetics with Osmotic Demyelination Syndrome: A Case Series and Literature Review.

Osmotic demyelination syndrome (ODS) is well known to be associated with the rapid correction of hyponatremia. However, there is limited literature on its link to persistent or corrected hyperglycemic states in diabetic patients. We report two young patients with ODS and hyperglycemia. Neuroimaging and blood tests indicated central pontine and extrapontine myelinolysis with a hyperglycemic hyperosmolar state (HHS). These unusual cases provide insight into the neurologic complications of hyperglycemia.

Interpreting mono- and poly-SCRA intoxications from an activity-based point of view: JWH-018 equivalents in serum as a comparative measure.

Synthetic cannabinoid receptor agonists (SCRAs) are a class of synthetic drugs that mimic and greatly surpass the effect of recreational cannabis. Acute SCRA intoxications are in general difficult to assess due to the large number of compounds involved, differing widely in both chemical structure and pharmacological properties. The rapid pace of emergence of unknown SCRAs hampers on one hand the timely availability of methods for identification and quantification to confirm and estimate the extent of the SCRA intoxication. On the other hand, lack of knowledge about the harm potential of emerging SCRAs hampers adequate interpretation of serum concentrations in intoxication cases. In the present study, a novel comparative measure for SCRA intoxications was evaluated, focusing on the cannabinoid activity (versus serum concentrations), which can be measured in serum extracts with an untargeted bioassay assessing ex vivo CB1 activity. Application of this principle to a series of SCRA intoxication cases (n = 48) allowed for the determination of activity equivalents, practically entailing a conversion from different SCRA serum concentrations to a JWH-018 equivalent. This allowed for the interpretation of both mono- (n = 34) and poly-SCRA (n = 14) intoxications, based on the intrinsic potential of the present serum levels to exert cannabinoid activity (cf. pharmacological/toxicological properties). A non-distinctive toxidrome was confirmed, showing no relation to CB1 activity. The JWH-018 equivalent was partly related to the poison severity score (PSS) and causality of the clinical intoxication elicited by the SCRA. Altogether, this equivalent concept allows to comparatively and timely interpret (poly-)SCRA intoxications based on CB1 activity.

Robotic spinal angiography: A single-center experience.

Robotic neurointervention enhances procedural precision, reduces radiation risk, and improves care access. Originally for interventional cardiology, the CorPath GRX platform has been used in neurointerventions. Recent studies highlight robotic cerebral angiography benefits, but information on spinal angiography is limited. While a new generation of robotic solutions is on the horizon, this series evaluates our experience with the CorPath GRX in spinal angiographic procedures, addressing a key gap in neurointerventional research. In this single-center retrospective case series, we analyzed 11 patients who underwent robotic-assisted diagnostic procedures with the CorPath GRX system from February 2022 to March 2023 at our institution. A descriptive synthesis was performed on the demographic, baseline, surgical, and postoperative data collected. The average age of the 11 patients was 54 ± 20.34 years, with six (54.55%) female. The mean body mass index was 29.58 ± 7.86, and 7 (63.64%) were non-smokers. Of the 11 procedures using the CorPath GRX system, four (36.36%) were partially converted to manual technique. General anesthesia was used in nine cases (81.82%), and right-side femoral access in ten (90.91%) patients. Mean fluoroscopy time was 24.81 ± 10.19 min, contrast dose 174.09 ± 57.31 mL, dose area product 472.23 ± 437.57 Gy·cm², and air kerma 2438.84 ± 2107.06 mGy. No robot-related complications and minimal procedure-related complications were reported. The CorPath GRX system, a robotic-assisted platform, has proven reliable and safe in spinal angiography, evidenced by its enhanced procedural accuracy and reduced radiation exposure for operators.

Pure arterial malformation of the fetal PCA treated with flow diverter stent-case report and literature review.

Thirty-three-year-old female underwent diagnostic work-up for chronic headaches. Cerebral DSA on the right side showed fetal posterior cerebral artery (PCA) with unusually shaped arterial malformation on its proximal part. Malformation was formed by multiple convolutions and loops with no arteriovenous shunting. During endovascular procedure, flow diverter stent was placed in the right C6 segment. Patient was put on double antiaggregation therapy. Four weeks after procedure, patient was admitted to ER with signs of dyphasia. No recent ischemic lesions were detected on emergent magnetic resonance. Dyphasia resolved spontaneously. On follow-up magnetic resonance angiography 5 years after the procedure, main portion of malformation has diminished with slightly filling of posterior part supplied by right P1. Here, reported finding is consistent with previously described pure arterial malformation (PAM). Main intention of our treatment was to induce malformation remodeling with flow diverter placement. Two main risks arise when using flow diverters on fetal PCA region. Firstly, there is possibility of occluding fetal PCA alongside malformation. On the contrary, high flow through fetal PCA could hinder malformation thrombosis. Some case series reported high rate of fetal PCA occlusion without ischemic sequelae in the PCA teritory. Others consider flow diverters highly ineffective treatment for the fetal PCA aneurysms due to low rate of complete aneurysm occlusion. We presented first reported case in which flow diverter was used to treat PAM. Slow malformation thrombosis leaves enough time for collateral circulation to recruit and supply territory of the vessel affected by the malformation.

Development and implementation of evidence-based, nurse-leading early warning model and healthcare quality improvement project for transplant-associated thrombotic microangiopathy: a mixed-methods, before-and-after study

ObjectiveThe early identification and diagnosis of transplant-associated thrombotic microangiopathy (TA-TMA) are essential yet difficult in patients underwent hematopoietic stem cell transplantation (HSCT). To develop an evidence-based, nurse-leading early warning model for TA-TMA, and implement the healthcare quality review and improvement project.MethodsThis study was a mixed-methods, before-and-after study. The early warning model was developed based on quality evidence from literature search. The healthcare quality review and improvement project mainly included baseline investigation of nurse, improvement action and effectiveness evaluation. The awareness and knowledge of early parameter of TA-TMA among nurses and the prognosis of patients underwent HSCT were compared before and after the improvement.ResultsA total of 1 guideline, 1 evidence synthesis, 4 expert consensuses, 10 literature reviews, 2 diagnostic studies, and 9 case series were included in the best evidence. The early warning model including warning period, high-risk characteristics and early manifestation of TA-TMA was developed. The improvement action, including staff training and assessment, suspected TA-TMA identification and patient education, was implemented. The awareness and knowledge rate of early parameter of TA-TMA among nurses significantly improved after improvement action (100% vs. 26.7%, P < 0.001). The incidence of TA-TMA was similar among patients underwent HSCT before and after improvement action (2.8% vs. 1.2%, P = 0.643), while no fall event occurred after improvement action (0 vs. 1.2%, P < 0.001).ConclusionThe evidence-based early warning model and healthcare quality improvement project could enhance the awareness and knowledge of TA-TMA among healthcare providers and might improve the prognosis of patients diagnosed with TA-TMA.

Dengue and leptospirosis coinfection: a case series

BackgroundBoth dengue and Leptospira infections are endemic to tropical and subtropical regions, with their prevalence increasing in recent decades. Coinfection with these pathogens presents significant diagnostic challenges for clinicians due to overlapping clinical manifestations and laboratory findings. This case report aims to elucidate two clinical scenarios where the coinfection of dengue and leptospirosis complicates the disease course, creating a diagnostic conundrum.Case presentationWe present the clinical scenarios of two Bangladeshi males, aged 25 and 35 years, who were admitted to our hospital with acute febrile illness. The first patient exhibited hepatic and renal involvement, while the second presented with symptoms initially suggestive of meningoencephalitis. Both cases were initially managed under the presumption of dengue infection based on positive serology. However, further evaluation revealed coinfection with Leptospira, complicating the disease course. Both patients received appropriate treatment for dengue and antibacterial therapy for leptospirosis, ultimately resulting in their recovery.ConclusionThese case scenarios underscore the critical importance for clinicians in regions where dengue and Leptospira are endemic to consider both diseases when evaluating patients presenting with acute febrile illness.

Evaluation of presentation, treatment and outcome in dogs with granulomatous steatitis associated with hypercalcaemia: six cases (2019-2023).

To report the presentation, treatment and outcome of dogs with granulomatous steatitis associated with total and ionised hypercalcaemia. Six dogs diagnosed with ionised and/or total hypercalcaemia and histologically diagnosed granulomatous steatitis were evaluated to determine the clinical signs, clinical findings, response to treatment and outcome. These cases were seen at different primary care and referral veterinary hospitals in the United Kingdom between 2019 and 2023. No alternative aetiology to explain the total and/or ionised hypercalcaemia or steatitis was identified. The most common presenting signs were lethargy, anorexia or hyporexia, vomiting and polyuria/polydipsia. Other clinical signs included weight loss, discomfort and panting. Five out of the six dogs responded to prednisolone. Four dogs were alive at the time of writing, one dog was lost to follow-up and one dog died 2 weeks post-diagnosis. It is well-established that granulomatous disease can cause hypercalcaemia. In this case series we found granulomatous steatitis associated with total and/or ionised hypercalcaemia. Dogs diagnosed with granulomatous steatitis should have ionised calcium measured, which may prompt further diagnostics and treatment options. Dogs with hypercalcaemia should be evaluated for evidence of steatitis where more common differentials have been excluded.

Adjunctive Intranasal Esketamine for Comorbid Treatment-Resistant Depression with Suicidal Ideation in Patients Receiving Palliative Care at a Comprehensive Cancer Center: A Case Series.

Background: The psychiatric needs of those with cancer and other advanced illnesses are becoming increasingly recognized. Ketamine is emerging as a promising treatment option for depressive disorders in psychiatric and palliative care. In the palliative care setting, its study has been hindered by lack of consistent administration routes and dosing. Intranasal (IN) esketamine (Spravato®) has recently received U.S. Food and Drug Administration (FDA) approval as an adjunctive agent for treatment-resistant depression (TRD) and major depressive disorder (MDD) with suicidal ideation (SI). Objective: We sought to offer IN esketamine to patients suffering from TRD and SI at a comprehensive cancer center. Methods: We designed and implemented a protocol to administer IN esketamine and describe three cases in which it was provided to patients with TRD and SI at a palliative care clinic in the United States. Results: Following treatment, all three patients had substantial reduction in depression severity and no further suicidalideation. These improvements were maintained for up to a year. No serious adverse events occurred. Conclusions: These cases illustrate the potential utility of IN esketamine in the palliative care setting.

Transobturator-cable-fixation in pelvic ring injuries with symphyseal disruption - a last resort?

The role of transobturator-cable-fixation (TOCF) in traumatic symphyseal rupture of the pelvic ring remains unclear. This case series aims to evaluate TOCF in complex and revision cases in pelvic surgery. A retrospective analysis of a chronological case series was conducted, studying pelvic fractures stabilized using TOCF between January 2006 and December 2022. The variables considered included age, gender, fracture classification, Injury Severity Score (ISS), Body Mass Index (BMI), trauma mechanism, time to surgery, fixation technique, hospital duration, complications, status on discharge (Glasgow Outcome Scale; GOS), follow-up time and indication for the use of TOCF. All patients (N = 7) were male with a mean age of 64 years and a mean BMI of 29. The mean ISS was 45, with the lowest ISS of 25, indicating that only polytraumatized patients were included. Two anterior-posterior-compression-, four lateral-compression-, and one vertical-shear-pelvic-injury were identified. TOCF was added in six cases to support symphyseal plating and in one case to external fixation. The mean hospital stay was 49 days and the mean follow-up duration was 8.5 months. No complications associated with TOCF were observed during the surgical procedure or follow-up. TOCF showed no procedure-associated complications and effectively supported symphyseal healing in all cases. The main indications were obesity, poor bone quality in elderly patients, and revision cases. TOCF could be considered as a last treatment option in open-book pelvic injuries where plating or external fixation is at risk to fail.

Basophil FceRI expression-A management tool in anti-IgE treatment of allergic asthma.

Immune-based therapy targeting immunoglobulin E (IgE), anti-IgE treatment, has emerged as an adjunct treatment for children with severe allergic asthma. After start of anti-IgE treatment, an effect of the treatment cannot be monitored by Total-IgE, because current methods measure both bound and free IgE molecules. Basophil activation test may be very useful for monitoring anti-IgE treatment efficacy. The objective of this paper is to evaluate if basophil activation test is applicable in regulating the anti-IgE treatment. A case series of 20 children with IgE-mediated severe allergic asthma were treated according to guidelines with anti-IgE (Omalizumab). Blood samples were drawn for total IgE, specific IgE, number of IgE receptors (FcεRI) and basophil sensitivity were measured at baseline before anti-IgE treatment and 4 months after initiation of anti-IgE treatment. A total of 19 out of 20 children had statistically significant and clinically relevant effects of anti-IgE treatment on symptom score, lung function and medication. All 20 children had a significant reduction in basophil allergen sensitivity and the number of IgE receptors (FcεRI) on blood basophils. Anti-IgE treatment was found to be well controlled by measuring basophil allergen sensitivity and FceRI density on blood basophils. This cohort study demonstrates a promising method, measuring basophil allergen sensitivity and in particular blood basophil FceRI density, concerning the monitoring of anti-IgE treatment in different clinical situations. There are no randomized controlled trials evaluating this method in clinical settings.

Emerging pathogens Aerococcus urinae and Aerococcus sanguinicola from a Canadian tertiary care hospital.

Background: Aerococcus urinae and Aerococcus sanguinicola are emerging pathogens linked with urinary tract infections. We present a case series of A. urinae and A. sanguinicola isolates characterizing the spectrum of clinical presentation, microbiological characteristics and antimicrobial sensitivities. Methods: Retrospective chart review was performed on patients who grew positive cultures for A. urinae and A. sanguinicola identified on MALDI-TOF in Saskatchewan from January to June 2023. Demographic and clinical variables, antimicrobial susceptibility and prescription were documented. Results: This cohort (n=115) had a median age 82 years. A. urinae and A. sanguinicola infections spanned from urinary tract infection (n=96) to urosepsis (n=6). These infections were predominantly monomicrobial (73.9%) and were susceptible to ceftriaxone, penicillin G and vancomycin. Antimicrobials were seldom prescribed within the urinary tract infection cohort (31.2%). Conclusion: Untreated A. urinae and A. sanguinicola infections can precipitate into urosepsis. The reported antimicrobial susceptibility for these Aerococcus isolates should be utilized to provide appropriate antimicrobial coverage.

Combination therapy consisting of transarterial chemoembolization, lenvatinib, and programmed cell death protein 1 blockade for hepatocellular carcinoma with inferior vena cava tumor thrombus: a case series study and literature review.

Patients with hepatocellular carcinoma (HCC) and inferior vena cava carcinoma tumor thrombus (IVCTT) have poor prognosis. Combination therapy involving blockade of programmed cell death protein 1 (PD-1) and tyrosine kinase inhibitors (TKIs) is an efficient treatment strategy for advanced HCC. However, surgical treatment after a combination of systemic therapy and transarterial chemoembolization (TACE) for HCC with IVCTT has not been widely reported, and the efficacy and safety of this treatment have not been studied. In the 21 cases reported herein, the patients were treated with TACE, lenvatinib, and PD-1 blockade. The treatment responses, progression-free survival (PFS), overall survival (OS), disease control rate, and toxicities were evaluated, and the related literature was reviewed. The overall response and disease control rates were 66.7% and 85.7%, respectively. The median PFS time was 16.0 months, with a 1-year PFS rate of 55.60%. The median OS was not reached, with a 1-year OS rate of 66.70%. Four patients underwent hepatectomy without serious complications and survived for 29.1, 24.7, 14.2, and 13.8 months. Three patients survived tumor-free, and one patient experienced intrahepatic recurrence. Pathological complete response and major pathological responses were observed in one and three patients, respectively. Treatment-related adverse events of any grade occurred in of 8/9 patients (88.9%), and grade 3 treatment-related adverse events occurred in one patient. The combination of TACE, lenvatinib, and PD-1 is effective for HCC with IVCTT and has acceptable adverse effects.

Keep it CooL! Results of a two-year CooL-intervention: a descriptive case series study

BackgroundCoaching on Lifestyle (CooL) is a two-year healthcare intervention for people with overweight or obesity, stimulating weight reduction by promoting sustained healthier behavior. The objective of this study is to investigate the effects of CooL on participants’ anthropometrics, personal factors and behavioral factors over the two-year timeframe of CooL.MethodsA descriptive case series study, using a broad set of routinely collected data on anthropometrics, personal factors and behavioral factors of adults living across the Netherlands. The data were collected between November 2018 and December 2021 among participants of CooL (N = 746) at three moments during the intervention: at baseline (T0), at 8 months (T1) after completion of phase 1 and at 24 months (T2) after ending CooL. Changes over time were analyzed using paired t-tests comparing baseline to T1 and baseline to T2. In addition, potential differences on outcomes in subgroups based on education level, weight status and group size were examined using paired t-tests and ANOVA-tests.ResultsThe results showed positive changes on all outcomes at 24 months compared to baseline. The largest effects were on perceived health, attentiveness towards meal size and meal composition (large effect size). Mean weight loss was 4.13 kg (SD 7.54), and mean waist circumference decreased with 4.37 centimeters (SD 8.59), indicating a medium to large effect size. Changes were consistent across subgroups varying in educational level, BMI at baseline and group size.ConclusionThe study demonstrated sustained weight-related effects of CooL over 24 months supporting its two-year duration. The results indicate that CooL, though not for every individual, is in general appropriate and effective for different group sizes and for a wide variety of participants regardless of level of education, or BMI at baseline.Trial registrationDutch Trial Register NTRNL6061 (13-01-2017). Registered at Overview of Medical Research in the Netherlands (OMON), via https://www.onderzoekmetmensen.nl/.

Audiovestibular outcomes in adult patients with cogan syndrome: a systematic review.

To determine factors associated with steroid responsiveness and efficacy of biologic disease-modifying anti-rheumatic (DMARD) use in patients with Cogan Syndrome (CS). A systematic search of Cochrane Library, PubMed, CINAHL, and Scopus was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines. Any study describing audiometric or vestibular data and pharmacologic treatment in patients with CS was included. Due to limited literature, only case reports/case series were included. Seventy case reports or case series studies comprising 79 individual cases of CS were included. A difference in vestibular symptoms with a higher prevalence in the steroid-resistant group than the steroid-responsive group was found (79.5% vs 57.9%, p = 0.04). Eighteen (60.0%) patients treated only with oral steroids had no audiological improvement, while twelve (n = 12; 85.7%) patients treated with biologic DMARD showed audiological improvement. The steroid-responsive group had an overall better response to DMARDs than the steroid-resistant group (62.1% vs 45.0%; 100.0% vs 77.8%). Our study synthesized the available literature to better characterize steroid resistance in patients with Cogan syndrome and treatment outcomes. Vestibular symptoms were noted to be more prevalent in patients who were eventually labeled as steroid resistant. There were higher rates of audiological improvement in patients given biologic DMARDs rather than conventional DMARDs or steroids only. Further studies are needed to characterize each individual vestibular symptom and investigate the utility and timing of biologic DMARDs in managing patients with Cogan syndrome.

Clinical features associated with immune checkpoint inhibitor nephritis: a single-center clinical case series

BackgroundAcute kidney injury (AKI) has been well described as a complication of immune checkpoint inhibitor therapy. We present a series of patients, the majority with lung adenocarcinoma, who developed AKI while actively receiving immune checkpoint inhibitors.MethodsThis is a retrospectively analyzed clinical case series of six patients treated at City of Hope Comprehensive Cancer Center. Data were collected on gender, age, ethnicity, comorbidities, concomitant medications, type of malignancy, treatments, and renal function. All patients underwent renal biopsy for classification of the mechanism of AKI. Comprehensive genomic profiling (CGP) was performed on tumor tissue for all patients.ResultsPatterns of AKI included acute interstitial nephritis and acute tubular necrosis. Contributing factors included the use of concomitant medications known to contribute to AKI. All but two patients had full resolution of the AKI with the use of steroids. There were several mutations found on CGP that was notable including an Exon 20 insertion as well as multiple NF1 and TP53 mutations. There was high PD-L1 expression on tumor tissue noted in two out of six patients. In addition to AKI, a subset of patients had proteinuria with biopsies revealing corresponding glomerular lesions of minimal change disease and focal and segmental glomerulosclerosis.ConclusionsOur case series demonstrates that AKI from immune checkpoint inhibitors has a variable presentation that may require an individualized treatment approach. Further studies are needed to identify biomarkers that may help identify those at risk and guide the management of this condition.

Advances in the multidisciplinary surgical approach to primary spinal sarcomas: insights from a retrospective case series on outcomes and survival

IntroductionThe management of spinal sarcomas is complex, given their widespread involvement and high recurrence rates. Despite consensus on the need for a multidisciplinary approach with surgery at its core, there is a lack of definitive guidelines for clinical decision-making. This study examines a case series of primary spinal sarcomas, focusing on the surgical strategies, clinical results, and survival data to inform and guide therapeutic practices.MethodsWe conducted a retrospective analysis of patients who underwent surgical resection for primary spinal sarcomas between 2005 and 2022. The study focused on gathering data on patient demographics, surgical details, postoperative complications, overall hospital stay, and mortality within 90 days post-surgery.ResultsThe study included 14 patients with a primary diagnosis of spinal sarcoma, with an average age of 48.6 ± 12.6 years. Chondrosarcoma emerged as the most common tumor type, representing 57.1% of cases, followed by Ewing sarcoma at 35.7%, and synovial sarcoma at 7.1%. Patients with chondrosarcoma were treated with en-bloc resection, while the patient with synovial sarcoma underwent intra-lesional excision and those with Ewing sarcoma received decompression and tumor debulking. Postoperative assessments revealed significant improvements in neurological conditions. Notably, functional status as measured by the Karnofski Performance Index (KPI), improved substantially post-surgery (from 61.4 to 80.0%) The mean follow-up was 34.9 ± 9.2 months. During this time period one patient experienced fatal bleeding after en-bloc resection complications involving the vena cava. None of the patient needed further surgery.ConclusionsOur 16-year study offers vital insights into managing primary spinal sarcomas, showcasing the effectiveness of surgical intervention, particularly en-bloc resection. Despite their rarity and complexity, our multidisciplinary treatment approach yields improved outcomes and highlights the potential for refined surgical strategies to become standardized care in this challenging domain.