Published Case Reports Research Articles

Animal and Human Dirofilariasis in India and Sri Lanka: A Systematic Review and Meta-Analysis.

Dirofilariasis is an emerging vector-borne tropical disease of public health importance that mainly affects humans and dogs. Dirofilaria immitis and D. repens are the two well-documented dirofilariasis-causing filarioid helminths of both medical and veterinary concerns in India and Sri Lanka. This systematic review and meta-analysis aimed to describe and summarize the current evidence of dirofilariasis prevalence and distribution in India and Sri Lanka. Interestingly, D. repens is reported to circulate in both dogs (prevalence of 35.8% (95% CI: 11.23-60.69)) and humans (97% of published case reports) in India and Sri Lanka, but D. immitis is reported to be present in the dog populations in India (prevalence of 9.7% (95% CI: 8.5-11.0%)), and so far, it has not been reported in Sri Lanka. This peculiar distribution of D. immitis and D. repens in the two neighbouring countries could be due to the interaction between the two parasite species, which could affect the pattern of infection of the two worm species in dogs and thus influence the geographical distribution of these two filarial worms. In medical and veterinary practice, histopathology was the most commonly used diagnostic technique (31.3%; 95% CI 2.5-60.2%). The low specificity of histopathology to speciate the various Dirofilaria spp. may lead to misdiagnosis. It was identified in this study that several regions of India and Sri Lanka have not yet been surveyed for dirofilariasis. This limits our understanding of the geographical distribution and interspecies interactions of the two parasites within these countries. Parasite distribution, disease prevalence, and interspecies interactions between the vectors and the host should be targeted for future research.

Management of traumatic fracture–dislocation of the shoulder in children: A systematic review of published case reports

Purpose Proximal humerus fracture occurring simultaneously with shoulder dislocation in children is extremely rare, with only a few reports of such cases having been reported. A systematic review of case reports was conducted to document outcomes after different treatment methods. Methods A systematic search of literature from 1980 to 2021 was performed from the PubMed/Medline, ScienceDirect, and SCOPUS databases. All case reports on traumatic fracture dislocation of the shoulder in children were included. Results A total of 18 studies were included with a mean follow-up of 1.5 years. There were 7 boys and 11 girls with an age range from 11 months to 16 years with a median of 6 years. These cases were grouped into two, based on the involvement of the epiphysis with fracture–dislocations. In group A, there were 11 cases (61%) of shoulder dislocation involving the epiphysis. While in group B, there were seven (38%) cases with fracture metaphysis but not involving the epiphysis. Both groups were treated by closed or open reduction with or without internal fixation. All fractures healed in a mean of 7 weeks while the full function was achieved in an average of 21.58 weeks. Conclusion This systemic review supports the treatment of patients in group A by open reduction with K-wire fixation. While in group B, closed reduction with intramedullary fixation is a better option as it provides satisfactory clinical and radiological results in a short time.

Optimizing the Safe Washout Period for Liver Transplantation Following Immune Checkpoint Inhibitors with Atezolizumab, Nivolumab, or Pembrolizumab

Using immune checkpoint inhibitors (ICIs) as a downstaging therapy for liver transplantation (LT) has improved outcomes for patients with advanced hepatocellular carcinoma (HCC). However, this therapy carries a risk of post-transplant graft rejection. The washout (WO) period between the last ICI dose and LT seems critical in preventing postoperative rejection. This study aimed to optimize the WO period by balancing tumor burden suppression and rejection prevention using ICIs before LT. We reviewed published case reports or series from March 2020 to December 2022 regarding LT for HCC after downstaging or bridge therapy with ICIs and included 4 of our cases. Most patients received atezolizumab, nivolumab, or pembrolizumab; these ICIs shared a half-life of around 28 days. Therefore, we excluded cases without definite WO period data and those using non-atezolizumab/nivolumab/pembrolizumab ICIs and ultimately enrolled 22 patients for analysis. We compared their clinical outcomes and estimated the rejection-free survival for every 0.5 half-life interval. Most study subjects received nivolumab (n=25). Six patients had severe rejections (nivolumab group, n=5) and needed rescue management. Of the 6 cases, 1 patient died after rejection, and 2 underwent re-transplantation. The median WO period in these 6 patients was 22 days (IQR: 9-35 days). In addition, we found that a 1.5 half-life (42 days) was the shortest safe WO period associated with significant rejection-free survival (P=.005). Our results showed that 42 days was the safest WO period before LT for HCC after ICI with atezolizumab, nivolumab, or pembrolizumab.

Adult-onset leukoencephalopathy with axonal spheroids and pigmented glia (ALSP) is commonly misdiagnosed as Frontotemporal Dementia (FTD), Multiple Sclerosis (MS,) Alzheimer’s disease (AD), or other adult-onset leukodystrophies (P12-4.003)

<h3>Objective:</h3> Characterize the misdiagnosis of ALSP <h3>Background:</h3> ALSP is a progressive, fatal autosomal dominant neurodegenerative disease caused by <i>CSF1R</i> gene mutations resulting in microglial dysfunction and profound white matter changes. ALSP prevalence is estimated at 10,000 in the US alone. The phenotype associated with ALSP is cognitive and motor impairment and can be misdiagnosed as FTD, MS or AD. Diagnostic accuracy of ALSP has modestly improved due to published diagnostic criteria. However, the rate of initial ALSP misdiagnosis remains high and involves a spectrum of neurodegenerative and neuroimmune disorders. <h3>Design/Methods:</h3> A systematic literature review of ALSP clinical and genetic features was conducted with published case studies (1/1/1980–3/22/2022). Data were extracted for 292 ALSP patients from 90 published case reports. Categorical variables were stratified by initial diagnosis, dichotomized by ALSP versus misdiagnosis categories and presented as frequency distribution. P-values for differences in frequency distribution between stratification factors were generated by Chi-square test. <h3>Results:</h3> Demographic data revealed the mean (SD) age (years) of patients was 43.2 (11.6), median (minimum, maximum) of 42.0 (18.0, 86.0). Family history of ALSP was identified in 58.9% of patients. Due to phenotypic heterogeneity at disease onset, accurate initial diagnosis was detected in only 31.5% of ALSP patients. FTD (11.6%), MS (7.9%), AD (4.5%), adult-onset leukodystrophy (6.8%), and familial leukoencephalopathy (6.8%) were common misdiagnoses. Cognitive impairment (45.9%) and behavioral and psychiatric dysfunction (26.4%) were the most common initial symptoms. <h3>Conclusions:</h3> This review confirms ALSP is commonly misdiagnosed as a cognitive disorder, AD/FTD, MS or other leukodystrophy. The presenting symptoms of ALSP family history, white matter MRI lesions and early-onset cognitive impairment with behavioral and/or motor dysfunction should trigger suspicion for ALSP and initiate genetic testing. To avoid misdiagnosis, screening patients for ALSP should be considered. Accurate diagnosis can allow for early intervention with investigational therapies currently in development. <b>Disclosure:</b> Dr. Meier has received personal compensation for serving as an employee of Vigil Neuroscience. Dr. Meier has received personal compensation for serving as an employee of Voyager Therapeutics. Dr. Meier has stock in Vigil Neuroscience. Dr. Meier has stock in Voyager Therapeutics. Dr. Leahy has received personal compensation in the range of $50,000-$99,999 for serving as a Consultant for Vigil Neuroscience. Dr. Leahy has stock in Ovid Therapeutics. Mrs. Pontius has received personal compensation for serving as an employee of Vigil Neuroscience, Inc. Mrs. Zappia has received personal compensation for serving as an employee of Vigil Neuroscience, Inc.. Mr. Brennan has received personal compensation for serving as an employee of Vigil Neuroscience. Dr. Papapetropoulos has received personal compensation for serving as an employee of Vigil Neuroscience . Dr. Papapetropoulos has received personal compensation in the range of $100,000-$499,999 for serving as a Consultant for Acadia. Dr. Papapetropoulos has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for Ventus Therapeutics Inc. Dr. Papapetropoulos has received personal compensation in the range of $10,000-$49,999 for serving as an officer or member of the Board of Directors for Adamas Pharmaceuticals. Dr. Papapetropoulos has received personal compensation in the range of $10,000-$49,999 for serving as an officer or member of the Board of Directors for Lipocine Inc. Dr. Papapetropoulos has stock in Vigil Neuroscience. Dr. Papapetropoulos has stock in Adamas. Dr. Papapetropoulos has stock in Lipocine. Dr. Wszolek has received personal compensation in the range of $5,000-$9,999 for serving as an Editor, Associate Editor, or Editorial Advisory Board Member for Polish Neurological Society/Via Medica.

Can we trust AI chatbots’ answers about disease diagnosis and patient care?

Background: Several chatbots that utilize large language models now exist. As a particularly well-known example, ChatGPT employs an autoregressive modeling process to generate responses, predicting the next word based on previously derived words. Consequently, instead of deducing a correct answer, it arranges the most frequently appearing words in the learned data in order. Optimized for interactivity and content generation, it presents a smooth and plausible context, regardless of whether the content it presents is true. This report aimed to examine the reliability of ChatGPT, an artificial intelligence (AI) chatbot, in diagnosing diseases and treating patients, how to interpret its responses, and directions for future development.Current Concepts: Ten published case reports from Korea were analyzed to evaluate the efficacy of ChatGPT, which was asked to describe the correct diagnosis and treatment. ChatGPT answered 3 cases correctly after being provided with the patient’s symptoms, findings, and medical history. The accuracy rate increased to 7 out of 10 after adding laboratory, pathological, and radiological results. In one case, ChatGPT did not provide appropriate information about suitable treatment, and its response contained inappropriate content in 4 cases. In contrast, ChatGPT recommended appropriate measures in 4 cases.Discussion and Conclusion: ChatGPT’s responses to the 10 case reports could have been better. To utilize ChatGPT efficiently and appropriately, users should possess sufficient knowledge and skills to determine the validity of its responses. AI chatbots based on large language models will progress significantly, but physicians must be vigilant in using these tools in practice.

Real-world efficacy of anti-IL-5 treatment in patients with allergic bronchopulmonary aspergillosis

Despite standard treatment with systemic corticosteroids and/or antifungal triazoles, a substantial proportion of patients with allergic bronchopulmonary aspergillosis (ABPA) experience frequent relapses and require long-term treatment despite unfavorable adverse effects. We investigated the efficacy and safety of anti-interleukin (IL)-5/IL-5 receptor α chain (Rα) monoclonal antibodies (mAbs) in patients with ABPA complicated by asthma. ABPA cases treated with anti-IL-5/IL-5Rα mAbs were collected from 132 medical institutes in 2018 and published case reports in Japan. Clinical outcomes, laboratory and physiological data, and radiographic findings during 32 weeks before and after treatment were retrospectively evaluated. We analyzed 29 cases of ABPA: 20 treated with mepolizumab and nine with benralizumab. Treatment with anti-IL-5/IL-5Rα mAbs reduced the frequency of exacerbations (p = 0.03), decreased the dose of oral corticosteroids (p < 0.01), and improved pulmonary function (p = 0.01). Mucus plugs in the bronchi shrank or diminished in 18 patients (82%). Despite the clinical/radiographical improvement, serum levels of total IgE, the key biomarker for the pharmacological response in ABPA, were unchanged. Anti-IL-5/IL-5Rα mAbs that directly target eosinophils are promising candidates for the treatment of patients with ABPA, especially those with mucus plugs in the bronchi.

Diagnostic Imaging for Distal Extremity Injuries in Direct Access Physical Therapy: An Observational Study.

Military physical therapists practicing direct-access routinely utilize diagnostic imaging and numerous published case reports demonstrate the ability of physical therapists to diagnose and appropriately disposition patients with foot/ankle and wrist/hand fractures. However, no larger cohort studies have explored the utilization of diagnostic imaging by physical therapists to detect fractures. To describe the utilization of diagnostic imaging in foot/ankle and wrist/hand injuries by physical therapists in a direct-access sports physical therapy clinic. Retrospective cohort study. The Agfa Impax Client 6 image viewing software (IMPAX) was searched from 2014 to 2018 for patients with diagnostic imaging ordered for foot/ankle and wrist/hand injuries. The Armed Forces Health Longitudinal Technology Application (AHLTA) electronic medical record was independently reviewed by the principal and co-investigator physical therapists. Data extracted were demographics and elements from the patient history and physical examination. In foot/ankle injuries, physical therapists diagnosed a fracture in 16% of the 177 cases and waited for an average of 3.9 days and 1.3 visits before ordering imaging. In wrist/hand injuries, physical therapists diagnosed a fracture in 24% of the 178 cases and waited for an average of 3.7 days and 1.2 visits before ordering imaging. The time to definitive care from the initial physical therapy evaluation was significantly different (p = 0.04) for foot/ankle fractures (0.6 days) compared to wrist/hand fractures (5.0 days). The Ottawa Ankle Rules demonstrated a negative likelihood ratio (-LR) of 0.11 (0.02, 0.72) and a positive likelihood ratio (+LR) of 1.99 (1.62, 2.44) for the diagnosis of foot/ankle fracture. Physical therapists utilizing diagnostic imaging in a direct-access sports physical therapy clinic diagnosed fractures in similar proportions for foot/ankle and wrist/hand injuries and quickly dispositioned patients to definitive care for those fractures. The diagnostic accuracy of the Ottawa Ankle Rules was similar to previously reported values. Level 3.

Solitary metachronous adrenal metastasis after radical resection of gastric large cell neuroendocrine carcinoma: a case report and literature review

Surgical resection is rarely employed for the treatment of metastatic gastric cancer, especially in patients with adrenal metastases, which usually indicate advanced systemic dissemination. Few published case reports have thus described the use of adrenalectomy for adrenal metastases from gastric cancer. In addition, most primary gastric malignancies are gastric adenocarcinomas, and gastric large cell neuroendocrine carcinoma (GLCNEC) is less common and has a poor prognosis. We report the case of a 71-year-old man who was diagnosed with solitary adrenal metastases 10 months after radical resection for GLCNEC and who was treated by adrenalectomy. He was followed-up for 9 months after adrenalectomy, with no further evidence of disease at his last follow-up examination. This case indicates that elective surgical resection may be feasible, even in rare cases of GLCNEC metastases to the adrenal glands, provided that the patient meets certain criteria, including solitary, metachronous tumors less than 4 cm.

Electroconvulsive Therapy in Thrombocytopenic Patients: A Case Report and Literature Review

AbstractIntroductionElectroconvulsive therapy (ECT) continues to be one of the most effective treatments for severe and treatment-resistant major depression. Although there are no absolute contraindications to performing ECT, the risks and benefits need to be assessed in all patients. We present a patient with a history of paroxysmal nocturnal hemoglobinuria (PNH) with significant thrombocytopenia, who completed an index course of six ECT sessions without complication. Currently, there are only two published case reports which describe the use of ECT in patients with thrombocytopenia.MethodsAD is a 37-year-old female with a history significant for MDD, GAD, panic disorder, PNH, and Budd Chiari, who presented to ED for suicidal ideation. Initial labs showed pancytopenia (Hb 4.56 mg/dl, Absolute Neutrophil 0.7 x 103/mL). Given her history of multiple failed SSRIs, ECT was proposed to treat her depression. Anesthesia delayed her initial pre-operative assessment due to concerns regarding her platelet count and risk of bleeding. They recommended transfusion with a preprocedural platelet count goal of 50 x 103/mL. Psychiatry recommended a platelet goal of 20 x 103/mL given previous ECT literature. Patient underwent six ECT sessions without complications. Her platelets ranged from 24–40 x 103/mL and she did not require preprocedural platelet transfusion.ResultsDue to a lack of published literature, there are no formal guidelines for performing ECT in thrombocytopenic patients. The first case report details a 64-year-old female who underwent 12 ECT sessions without complication, while her platelet count ranged from 7–38 x 103/mL. The most recent case report describes a 74-year-old male who underwent nine ECT treatments without complications. The authors decided to transfuse prophylactically if pre-procedure platelet count was less than 20 x 103/mL. The patient underwent nine treatments, requiring eight transfusions. CT head did not show any signs of hemorrhage or structural changes. The authors remarked it is unclear whether the transfusions were necessary.ConclusionsEven though ECT has been in use for over 80 years, there is still much that is unknown about this treatment modality. This case highlights the lack of absolute contraindications to performing ECT. However, the lack of literature and studies regarding platelet goals in thrombocytopenic patients for ECT delayed patient care in this specific case. We add a third case report in a thrombocytopenic patient who underwent ECT without complications with a pre-procedure transfusion criterion if platelet count was less than 20 x 103/mL. More research needs to be conducted to determine risk and cut-off limits for platelet transfusion prior to performing ECT in thrombocytopenic patients.FundingNo funding

Azathioprine-Induced Hypersensitivity Vasculitis

Vasculitic skin rash is a rare but known manifestation of azathioprine hypersensitivity reactions, with several published case reports. In this report, we describe the case of a 63-year-old man on azathioprine for autoimmune hepatitis, who developed a delayed systemic hypersensitivity reaction with biopsy-proven vasculitis, approximately 10 months into his treatment. This resolved after azathioprine discontinuation and has not recurred to date after subsequent administration of 6-mercaptopurine. This case highlights the need for continued monitoring for delayed hypersensitivity reactions to azathioprine after initiation of therapy.

5613019 THE CO-EXISTENCE OF SICKLE CELL DISEASE WITH NEUROFIBROMATOSIS: A CASE REPORT

Sickle Cell disease is an autosomal recessive disorder that occurs as a result of a point mutation in the haemoglobin beta gene found on chromosome 11p15. It affects approximately 1 in 2000 livebirths in the UK1. Neurofibromatosis Type 1 has an incidence of around 1:30002. Neurofibromatosis is caused by mutations in the NF1 gene found on chromosome 17. Although it can be inherited in an autosomal dominant fashion, in about 50 percent of individuals Neurofibromatosis occurs spontaneously2. We present the case of 10 year old girl of Caribbean descent who was born prematurely at 27 weeks gestation. She was diagnosed with Sickle Cell disease on routine newborn screening. In infancy she had chronic lung disease, poor growth, and developmental delay. Throughout childhood she has required numerous prolonged admissions with vaso-occlusive crisis and chest infections. Her clinical condition is prone to rapid decompensation and has frequently been transferred to intensive care for management of both significant chest infections and acute chest syndrome. Her growth continues to follow the 0.4th centile. She also has learning difficulties and features of autism. These complications were initially thought to be related to her prematurity and sickle cell disease. However, more recently she developed an increasing number of freckles to her torso. She has multiple café au lait patches with axillary and groin freckling, meeting the diagnostic criteria for Neurofibromatosis 1. This is likely to have occurred as a de novo mutation as no other family members are affected. Despite the common occurrence of both these genetic disorders, a literature search found there has only been one previous published case report worldwide of a patient with both Sickle Cell disease and Neurofibromatosis Type 13. In this case report we describe the patient’s clinical course and management to date, and consider the need to look for additional diagnoses in patients with multiple complications.

Epidemiology of Mucormycosis in Greece; Results from a Nationwide Prospective Survey and Published Case Reports.

Mucormycosis has emerged as a group of severe infections mainly in immunocompromised patients. We analysed the epidemiology of mucormycosis in Greece in a multicentre, nationwide prospective survey of patients of all ages, during 2005-2022. A total of 108 cases were recorded. The annual incidence declined after 2009 and appeared stable thereafter, at 0.54 cases/million population. The most common forms were rhinocerebral (51.8%), cutaneous (32.4%), and pulmonary (11.1%). Main underlying conditions were haematologic malignancy/neutropenia (29.9%), haematopoietic stem cell transplantation (4.7%), diabetes mellitus (DM) (15.9%), other immunodeficiencies (23.4%), while 22.4% of cases involved immunocompetent individuals with cutaneous/soft-tissue infections after motor vehicle accident, surgical/iatrogenic trauma, burns, and injuries associated with natural disasters. Additionally, DM or steroid-induced DM was reported as a comorbidity in 21.5% of cases with various main conditions. Rhizopus (mostly R. arrhizus) predominated (67.1%), followed by Lichtheimia (8.5%) and Mucor (6.1%). Antifungal treatment consisted mainly of liposomal amphotericin B (86.3%), median dose 7 mg/kg/day, range 3-10 mg/kg/day, with or without posaconazole. Crude mortality was 62.8% during 2005-2008 but decreased significantly after 2009, at 34.9% (p = 0.02), with four times fewer haematological cases, fewer iatrogenic infections, and fewer cases with advanced rhinocerebral form. The increased DM prevalence should alert clinicians for timely diagnosis of mucormycosis in this patient population.

A Systematic Review of Research Developments in Mass Psychogenic Illness

This study explores the development of research on mass psychogenic illness (MPI) over recent decades. The literature review was used as the research methodology for this study, and dozens of published case reports and studies are reviewed in this paper. First, the paper reviews past researchers' general recognition of mass psychogenic illness. The paper includes the two subtypes of mass psychogenic disorders, 'mass anxiety hysteria' and 'mass motor hysteria', as well as the characteristics and common symptoms of the outbreak of mass psychogenic illness and the suspicion of the existence of mass psychogenic illness by some scholars. Secondly, historical cases of mass psychogenic illness outbreaks are reviewed in chronological order, from the Middle Ages through to the 21st century. Thirdly, new trends in the development of mass psychogenic illness and a new type of mass psychogenic illness, MSMI, proposed by researchers are reviewed. Finally, two important studies from the past decade are reviewed. The findings of two studies, the inducibility of mass psychogenic illness and hypnotizability, an important predictor to the emergence of cases of mass psychogenic illness, are also presented. Besides, some implications for future research into mass psychogenic illness are given through the review.

Vulval Aphthous Ulcers in Adolescents Following COVID-19 Vaccination—Analysis of an International Case Series

To review and characterize reports of vulval aphthous ulcers (VAUs) following COVID-19 vaccination in VigiBase, the World Health Organization global database of reported potential adverse effects of medicinal products, to demonstrate the importance and power of case reports for rare suspected adverse reactions and to investigate whether they suggest a potential for COVID-19 vaccination to be a trigger METHODS: Cases reporting the Medical Dictionary for Regulatory Activities' (MedDRA) preferred term "Vulvovaginal ulceration" and related preferred terms in adolescent patients aged 12-17 years in association with any COVID-19 vaccine were extracted from VigiBase. The cases were clinically reviewed, and causality was assessed by applying the Bradford Hill criteria to the obtained case series. As of June 30th, 2022, there were 444 reports for the selected Medical Dictionary for Regulatory Activities preferred terms following COVID-19 vaccination in VigiBase. Ninety-four de-duplicated reports concerned adolescent female patients. Thirty-seven cases were clinically consistent with the diagnosis of VAU. On causality assessment, the analyzed case series fulfilled 6 of the 9 Austen Bradford Hill criteria supporting a potential causal relationship. VAUs can be perceived as a traumatic experience, especially in adolescent patients. There is, furthermore, a risk that the ulcers will be misdiagnosed, resulting in avoidable investigation and treatment burdens for patients. We communicate our findings to support the small number of published case reports and raise awareness of VAUs occurring in a temporal association with COVID-19 vaccination. Furthermore, our analysis supports observations about the value of case reports for the recognition and assessment of rare adverse events.

Pneumocystis jirovecii pneumonia associated with immune checkpoint inhibitors: A systematic literature review of published case reports and disproportionality analysis based on the FAERS database.

Background: Pneumocystis jirovecii pneumonia (PJP) has been reported with ICIs but limited to case reports. The clinical features of PJP with ICIs remain mostly unknown. This study aims to investigate the association of PJP with ICIs and describe clinical features. Methods: Reports of PJP recorded in FAERS (January 2004-December 2022) were identified through the preferred term "Pneumocystis jirovecii pneumonia". Demographic and clinical features were described, and disproportionality signals were assessed through the Reporting Odds Ratio (ROR) and Information Component (IC), using traditional chemotherapy and targeted therapy as comparators, and adjusting signals by excluding contaminant immunosuppressive drugs and pre-existing diseases. A systematic literature review was conducted to describe clinical features of published PJP reports with ICIs. Bradford Hill criteria was adopted for global assessment of the evidence. Results: We identified 677 reports of PJP associated with ICIs, in which 300 (44.3%) PJP cases with fatal outcome. Nivolumab (IC025 2.05), pembrolizumab (IC025 1.88), ipilimumab (IC025 1.43), atezolizumab (IC025 0.36), durvalumab (IC025 1.65), nivolumab plus ipilimumab (IC025 1.59) have significant signals compared to other drugs in FAERS database. After excluding pre-existing diseases and immunosuppressive agents which may increase susceptibility of PJP, the signals for PJP associated with nivolumab, pembrolizumab, durvalumab, nivolumab plus ipilimumab remained robust (IC025 > 0). When compared to other anticancer regimens, although all ICIs showed a lower disproportionate signal for PJP than chemotherapy, nivolumab (IC025 0.33, p < 0.001), pembrolizumab (IC025 0.16, p < 0.001), both PD-1 inhibitors, presented a higher signal for PJP than targeted therapy. Male gender (IC025 0.26, p < 0.001) and age >65years (IC025 0.38, p < 0.001) were predominant in PJP cases associated with across all ICIs. In literature, 15 PJP cases associated with ICIs were reported in 10 published case reports. 12 of 15 (80.0%) of cases received PD-1 inhibitors before PJP was diagnosed. Conclusion: By the combined analysis of post-marketing data from FAERS and published case reports, we identified ICIs may be associated with PJP, especially in males aged >65years. After accounting for confounders, PD-1 inhibitors emerged with a robust disproportionality signal when compared to PD-L1/CTLA-4 inhibitors as well as targeted therapy. Further research is warranted to validate our findings.

Emotional Lability as a Symptom of Extra-axial Posterior Fossa Tumors: a Case-Control Review of Neuroanatomy and Patient-Reported Quality of Life.

Introduction Emotional lability (EL), the uncontrollable and unmotivated expression of emotion, is a rare and distressing symptom of brainstem compression. In published case reports, EL from an extra-axial posterior fossa tumor was alleviated by tumor resection. The primary aim herein was to radiographically establish the degree of compression from mass lesions onto brainstem structures. Secondarily, we compared changes in patient-reported quality of life (QOL) pre- and postoperatively. Methods A retrospective review of posterior fossa tumors treated between 2002 and 2018 at Vancouver General Hospital revealed 11 patients with confirmed EL. Each case was matched to three controls. A lateral brainstem compression scale characterized mass effect at the level of the medulla, pons, and midbrain in preoperative axial T2-weighted fluid-attenuated inversion recovery magnetic resonance imaging (FLAIR MRI) scans. Compression and clinical variables were compared between patient groups. Short Form-36 version 1 health surveys were retrospectively obtained from patient charts to compare pre- versus postoperative changes in survey scores between EL and control patients. Results EL symptoms ceased postoperatively for all EL patients. EL tumors exert greater compression onto the pons ( p = 0.03) and EL patients more commonly have cerebellar findings preoperatively ( p = 0.003). Patients with EL-causing tumors experienced greater improvement postoperatively in "Health Change" ( p = 0.05), which was maintained over time. Conclusion Findings suggest that compression onto the pons inhibits control over involuntary, stereotyped expression of emotion. This adds to evidence that EL may be attributed to cerebellum deafferentation from cortical and limbic structures through the basis pontis, leading to impaired modulation of emotional response. QOL results augment benefits of offering patients EL-alleviating tumor resection surgery.

The ocular manifestations of relapsing polychondritis.

Relapsing polychondritis (RPC) is a rare, multi-system, inflammatory disorder. Ocular disease is estimated to occur in 14-67% of patients with RPC, and any ocular structure can be affected. Published case reports and series of RPC were analysed to determine the frequency and nature of the ocular manifestations of RPC, including the age and gender distribution. A literature search of the MEDLINE database for case reports and series on RPC was conducted in October 2021 using search terms [relapsing polychondritis (MeSH Major Topic)] OR [relapsing polychondritis (Title/Abstract)]. Articles were included if the diagnosis of RPC was confirmed using established diagnostic criteria and if the paper described the clinical features of patients with RPC. 546 articles (454 case reports and 92 case series) described the clinical features in a total of 2414 patients with RPC. 49% of patients with RPC had ocular involvement, and this was a presenting feature in 21%. The most common ocular manifestations were scleritis (32%), episcleritis (31%) and uveitis (23%). Many patients with RPC will be seen by an ophthalmologist during the course of their disease. Knowledge and awareness of RPC and its ocular manifestations is therefore essential to enable the ophthalmologist to make the diagnosis.

Donepezil-induced psychosis: a cautionary report of a rare adverse reaction

IntroductionDonepezil is an acetylcholinesterase inhibitor approved by the Food and Drug Administration for the treatment of dementia in Alzheimer’s disease. While it is not curative for Alzheimer’s disease, donepezil has been shown to improve symptoms and slow disease progression; however, cases of rare psychiatric adverse effects, including hallucinations, mania, and increased confusion, have been reported. This report presents a case of donepezil-induced psychosis, which quickly resolved following cessation of the offending medication.ObjectivesTo illustrate a unique case of donepezil-induced psychosisMethodsThe patient is an 81-year-old male with a history of late-onset Alzheimer’s disease, mild depression, hypertension, hyperlipidemia, gastroesophageal reflux disease, and myocardial infarction. The patient was prescribed oral donepezil 10mg twice daily to manage his late-onset Alzheimer’s disease. Subsequently, he began developing persecutory delusions, increased agitation toward his family, and auditory hallucinations. His symptoms were worse at night after taking the donepezil, and he regularly requested to have a firearm for self-defense in the late hours of the night. His symptoms progressed for several weeks before his family brought him into the geriatric psychiatry clinic to address his psychosis. The family recognized that these new symptoms started shortly after the patient began taking donepezil and had already started decreasing the dose to half of what was originally prescribed.ResultsThis patient experienced symptom remission from psychosis immediately upon discontinuation of donepezil. The patient and his family reported significant improvement with no continuation of hallucinations or paranoia. There was also reported improvement in mood and irritability, and the patient appeared significantly better upon follow-up with geriatric psychiatry. Due to this immediate improvement, the suspected causative factor in the precipitation of psychosis in this patient is the anticholinesterase activity of the donepezil. Although the prescribing information of donepezil details inadequate data proving an association between donepezil and psychotic symptoms, two other published case reports (Yorston GA et al. J Psychopharmacol 2000;14:303-4, Pozzi FE et al. Case Rep Neurol 2022; 14:359-365), along with this one, provide evidence of a causal relationship between the two. The patient was switched to memantine therapy and has remained free of psychotic symptoms thus far.ConclusionsThis case demonstrates the caution required among clinicians when prescribing donepezil for the treatment of Alzheimer’s disease. There needs to be a more focused risk evaluation of potential psychiatric adverse effects in patients treated with donepezil.Disclosure of InterestNone Declared

Dextrocardia y herniación intratorácica del hígado. Una rara entidad

Introduction: the incidence of dextrocardia as a congenital anomaly is less than 0.01% and the combination with intrathoracic herniation of the liver resembling a benign neoplasm without a history of open or blunt thoracoabdominal trauma makes it even less frequent. Clinical case: the case of a 34-year-old female patient who consults for back pain is presented. On physical examination, heart sounds are auscultated in the right hemithorax and the chest X-ray shows dextrocardia and an image that resembles a right supradiaphragmatic mass. Triphasic CT confirms the presence of a rounded protrusion of a segment of the liver through a non-open defect of the liver right hemidiaphragm. The treatment has been conservative. Conclusion: the combination of dextrocardia accompanied by herniation of a portion of the liver through a defect in the right diaphragm is an extremely rare association and published case reports are few.

The Pancreas in Coronavirus Disease 2019 Infection.

An association between acute pancreatitis (AP) and coronavirus disease 2019 (COVID-19) has been proposed but the mechanisms of pancreatic injury of the severe acute respiratory syndrome coronavirus-2 (SARS-CoV-2) and the implicative role on the development of AP are not yet fully understood. COVID-19 also imposed major challenges on pancreatic cancer management. We conducted an analysis on the mechanisms of pancreatic injury by SARS-CoV-2 and reviewed published case reports of AP attributed to COVID-19. We also examined the pandemic effect on pancreatic cancer diagnosis and management, including pancreatic surgery.