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BMJ Open Ophthalmology Jun 2024Graves' ophthalmopathy is a complex autoimmune disorder that can significantly affect quality of life (QoL), vision and physical appearance. Recently, a deeper...
BACKGROUND
Graves' ophthalmopathy is a complex autoimmune disorder that can significantly affect quality of life (QoL), vision and physical appearance. Recently, a deeper understanding of the underlying pathogenesis has led to the development of novel treatment options.
AIMS
The purpose of this review is to explore the current literature on conventional and novel treatment modalities and to evaluate which interventions provide the most favourable psychological and clinical outcomes in patients with moderate to severe, active Grave's ophthalmopathy. For example, QoL is an important psychosocial outcome of disease management. However, available literature demonstrates that not all clinically effective treatment options improve patients' QoL.
METHODS
A systematic literature review was conducted to assess the clinical and psychosocial outcomes of different therapies for Graves' ophthalmopathy. An extensive database search of Ovid Medline, Ovid Embase and Cochrane Central Register of Controlled Trials was conducted. Studies generated were reviewed and the relevant selected data were retrieved and analysed.
RESULTS
Results showed intravenous steroids, rituximab (RTX), tocilizumab and teprotumumab were all significantly effective in improving Clinical Activity Scores. Orbital radiotherapy showed a slight improvement in proptosis and diplopia. All interventions were safe with few serious adverse events being reported across all studies. All treatment modalities demonstrated beneficial improvements in both components of the Graves' Ophthalmopathy-QoL (QoL) questionnaire, apart from orbital radiotherapy which only demonstrated improvements in the visual functioning subscale. Teprotumumab was identified to be the most effective intervention for improving both clinical and psychosocial outcomes. However, further research needs to be conducted to evaluate its side effect profile and cost-effectiveness. Nonetheless, with time it has the potential to be a first-line treatment option in the management of active moderate to severe Graves' ophthalmopathy.
Topics: Humans; Graves Ophthalmopathy; Quality of Life; Antibodies, Monoclonal, Humanized; Rituximab; Immunologic Factors; Glucocorticoids
PubMed: 38886120
DOI: 10.1136/bmjophth-2023-001515 -
Nutrition Journal Jun 2024This meta-analysis aims to analyze the relationship between serum vitamin D (VD) levels and Graves' disease (GD). (Meta-Analysis)
Meta-Analysis
OBJECTIVE
This meta-analysis aims to analyze the relationship between serum vitamin D (VD) levels and Graves' disease (GD).
METHODS
We conducted a search for publications on VD and GD in the English language. Our search encompassed databases such as PubMed, Embase, Web of Science, and the Cochrane Library, covering publications available through August 2023. A meta-analysis was performed using Cochrane RevMan 5.4 software. The standardized mean difference (SMD) and 95% confidence interval (CI) were used for outcome calculation. We used R software to test for publication bias.
RESULTS
Twelve studies were selected, comprising 937 (22.4%) cases with GD and 3254 (77.6%) controls. The overall meta-analysis revealed that patients with GD are significantly more likely to have low VD levels (SMD = - 0.66; 95% CI: -1.05, - 0.27; p = 0.001) than those in the control group. Egger's test results indicated no publication bias (p = 0.0791). These studies exhibited a high degree of heterogeneity (chi-square = 205.86, p < 0.00001; I = 95%). Subgroup analysis was conducted based on assay method, geographic location, and mean age of the case group to explore the heterogeneity sources. Assay methods and geographic locations were identified as potential heterogeneity sources. Based on the mean age, there were no statistically significant differences found in the subgroup analysis of the included studies.
CONCLUSION
There is promising evidence that low serum VD levels may increase the risk of GD. Further rigorous and long-term trials are needed to explore the role of VD in the onset and treatment of GD.
Topics: Humans; Graves Disease; Vitamin D; Vitamin D Deficiency
PubMed: 38849834
DOI: 10.1186/s12937-024-00960-2 -
Eye (London, England) Jun 2024Addressing Dysthyroid Optic Neuropathy (DON) is crucial due to its debilitating impact in thyroid eye disease (TED). Prompt treatment can preserve vision. Despite... (Meta-Analysis)
Meta-Analysis
PURPOSE
Addressing Dysthyroid Optic Neuropathy (DON) is crucial due to its debilitating impact in thyroid eye disease (TED). Prompt treatment can preserve vision. Despite lacking definitive diagnostic criteria, computed tomography's (CT) parameters are commonly used for diagnosis. However, these parameters exist without consensus on their diagnostic performance.
DESIGN
Systematic review and meta-analysis.
METHODS
We conducted a meta-analysis of studies assessing orbital CT diagnostic performance for DON in adults with TED. We searched various databases including Medline, PubMed, Scopus, and EMBASE, and others electronic databases, until July 2023. Evaluated CT parameters includes Barrett index (BI), fat prolapse via superior-orbital-fissure (SOF), superior-ophthalmic-vein-dilatation (SOVD), and the Nugent score. Diagnostic Test Accuracy analysis (DTA) was performed using R.
RESULTS
A total of 9 articles with documented target parameters, collectively analysed 212 orbits with DON. Nugent score exhibited highest diagnostic ability with a log diagnostic odd ratio (logDOR) of 2.64 (95% CI, 2.02, 3.25). Another significant DON indicator was a BI ≥ 50%, with a logDOR of 1.97 (95% CI, 1.17; 2.77). Conversely, fat prolapse via SOF and SOVD proved less sensitive, with a logDOR of 1.42 and 1.09 respectively. Regarding the SROC curve, Nugent score and the BI have the greatest AUC. Variations in study locale, participant demographics, and measurement methods accounted for heterogeneity in meta-analysis.
CONCLUSIONS
Nugent score and a BI ≥ 50% prove to be significant diagnostic parameters for DON, distinguishing them from fat prolapse via SOF and SOVD. Prioritizing these parameters can lead to prompt treatment and thus enhanced visual outcomes.
PROSPERO REGISTRATION NUMBER
CRD42023446376.
Topics: Humans; Graves Ophthalmopathy; Tomography, X-Ray Computed; Optic Nerve Diseases; Orbit
PubMed: 38472378
DOI: 10.1038/s41433-024-03011-6 -
Arquivos Brasileiros de Oftalmologia 2024We aimed to study reported cases of nasopharyngeal carcinoma presenting with ophthalmic manifestations with and without a prior diagnosis of nasopharyngeal carcinoma.
PURPOSE
We aimed to study reported cases of nasopharyngeal carcinoma presenting with ophthalmic manifestations with and without a prior diagnosis of nasopharyngeal carcinoma.
METHODS
We conducted a systematic review following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA). A literature search was conducted using the MEDLINE database in PubMed and Google Scholar. We included patients with a previous diagnosis of nasopharyngeal carcinoma in Group I and those without a prior diagnosis of nasopharyngeal carcinoma in Group II. Data included demographics, clinical presentation, history of nasopharyngeal carcinoma, treatment, histopathological description, World Health Organization classification, and outcome.
RESULTS
Fifty-eight patients (26 in Group I and 32 in Group II) were included. The male-to-female ratio was 3:1. The mean age of the patients (53.3 ± 11.7 years and 54.8 ± 16.2 years, respectively) and gender did not differ significantly between the two groups. The most common ocular presentations were diplopia and proptosis in the first group (each in 34.6%), whereas visual disturbance was most common in the second group (46.9%). Treatment options and World Health Organization grading were comparable. The outcome in 38 patients (after a comparable follow-up period) was significantly better in group II (p=0.003). There was no statistically significant difference in the outcome of 23 patients in correlation with World Health Organization grades II versus III irrespective of group (p=0.094).
CONCLUSIONS
The demographics of patients with nasopharyngeal carcinoma presenting with ophthalmic manifestations were similar between the two study groups, with a wide age range and male predominance. Patients presenting initially to ophthalmologists with no history of nasopharyngeal carcinoma have a more favorable outcome. World Health Organization grading may have less value as a prognostic indicator.
Topics: Humans; Databases, Factual; Exophthalmos; Eye; Nasopharyngeal Carcinoma; Nasopharyngeal Neoplasms; Eye Diseases
PubMed: 38451680
DOI: 10.5935/0004-2749.2022-0241 -
Ophthalmic Plastic and Reconstructive... Feb 2024A primary orbital respiratory cyst is a congenital choristoma that presents in the orbit and with different signs and symptoms depending on the location, which might...
A primary orbital respiratory cyst is a congenital choristoma that presents in the orbit and with different signs and symptoms depending on the location, which might also change the surgical approach. The aim of this report is to describe 2 new cases of primary respiratory epithelial cysts and to review the literature on presentation, management, and risk factors with different surgical approaches and complications. Two cases presenting with gradually increased proptosis had a confirmed diagnosis of a respiratory epithelial cyst. CT and MRI imaging revealed a thin-walled intraconal cystic lesion. Complete surgical removal was not possible and an incisional biopsy of the wall was performed. Rarely, orbital respiratory epithelium cysts are primary lesions. The high risk of breaking the capsule during surgery and the presence of residual epithelial cells within the orbit may cause cyst's recurrence. Indeed, we recommend careful attention during dissection maneuvers and a long-term follow-up.
PubMed: 38427820
DOI: 10.1097/IOP.0000000000002647 -
Biomedicines Feb 2024The ocular involvement of neuroendocrine neoplasms (NENs) is uncommon and mainly represented by metastases from gastrointestinal and lung neuroendocrine tumors. Primary... (Review)
Review
BACKGROUND
The ocular involvement of neuroendocrine neoplasms (NENs) is uncommon and mainly represented by metastases from gastrointestinal and lung neuroendocrine tumors. Primary orbital NENs are even less common and their diagnostic and therapeutic management is a challenge.
METHODS
A systematic review of the literature was conducted from 1966 to September 2023 on PubMed to identify articles on orbital NENs and to summarize their clinical-pathological features, diagnosis and therapeutic management. Furthermore, we presented a case of a locally advanced retro-orbital primary neuroendocrine tumor that was referred to the certified Center of Excellence of Sant'Andrea Hospital, La Sapienza University of Rome, Italy.
RESULTS
The final analysis included 63 records on orbital NENs and 11 records focused on primary orbital NENs. The localization was mostly unilateral and in the right orbit; proptosis or exophthalmos represented the initial symptoms. The diagnostic work-up and therapeutic management was discussed and a diagnostic algorithm for the suspicion of primary orbital NENs was proposed.
CONCLUSIONS
A multidisciplinary approach is required for the management of primary orbital NENs, emphasizing the importance of early referral to dedicated centers for prompt differential diagnosis, tailored treatment, and an improved quality of life and survival.
PubMed: 38397981
DOI: 10.3390/biomedicines12020379 -
BMC Endocrine Disorders Jan 2024To evaluate the efficacy and safety of combined glucocorticoids (GCs) and cyclophosphamide (CYC) treatment in Graves' ophthalmopathy (GO). (Meta-Analysis)
Meta-Analysis
PURPOSE
To evaluate the efficacy and safety of combined glucocorticoids (GCs) and cyclophosphamide (CYC) treatment in Graves' ophthalmopathy (GO).
METHODS
We searched PubMed, Embase, Cochrane Library, and four Chinese databases (Chinese National Knowledge Infrastructure (CNKI), China Science and Technology Journal Database (VIP), WanFang, and SinoMed) for any published randomized controlled trials (RCTs) produced from inception to December 1, 2023. Articles obtained using appropriate keywords were selected independently by two reviewers according to the established inclusion and exclusion criteria.
FINDINGS
We retrieved 1120 records which were eventually reduced to 13 RCTs which were then included in this evaluation. Pooled results indicated that the experimental group (CYC/GCs) showed a higher response rate than control group (GCs or negative control) (RR 1.27; 95% confidence interval 1.19 to 1.37). The subgroup analysis showed that the difference in response rates among treatment protocols (CYC/P, CYC/MPS, CYC/DEX) was not statistically significant (p = 0.23).
IMPLICATIONS
The combination of GCs and CYC could be recommended as a therapeutic option for GO, especially in patients who experience recurrence after a withdrawal GCs, have a poor response to GCs, or cannot obtain monoclonal antibody agents for various reasons.
Topics: Humans; Glucocorticoids; Graves Ophthalmopathy; Cyclophosphamide; China
PubMed: 38273269
DOI: 10.1186/s12902-024-01545-0 -
Ophthalmic Plastic and Reconstructive... Jan 2024To review existing literature concerning the effectiveness and safety of tocilizumab (TCZ) for managing thyroid eye disease.
PURPOSE
To review existing literature concerning the effectiveness and safety of tocilizumab (TCZ) for managing thyroid eye disease.
METHODS
A systematic search was conducted across the PubMed and Embase databases to identify studies on TCZ therapy, from inception to May 2023. The search included the keywords "Graves orbitopathy," "thyroid ophthalmopathy," "thyroid eye disease," "thyroid-associated orbitopathy," "thyroid-associated ophthalmopathy," "Graves ophthalmopathy," "endocrine ophthalmopathy," and "Tocilizumab." Only articles written in English, Spanish, or French were considered.
RESULTS
Among the 1,013 articles initially screened, a total of 29 fulfilled the eligibility criteria and were selected. Most studies were case reports or case series, and only one randomized clinical trial was found. TCZ has been used mainly in glucocorticoid-resistant or relapsing cases, with a dosage ranging from 4 or 8 mg/kg every 4 weeks when intravenous or a weekly subcutaneous dose of 162 mg. Treatment duration is usually adjusted to the clinical response. TCZ is mostly effective in reducing inflammatory signs during the active phase of thyroid eye disease, with an improvement of at least 3 points in clinical activity score and an overall relapsing rate of 8.2%. Numerous studies have shown marked reductions in proptosis; although the only available randomized controlled trial reported a nonstatistically significant improvement 6 months after treatment, a recent meta-analysis indicated that TCZ seems to be the most effective treatment for reducing proptosis. No severe side effects related to intravenous or subcutaneous TCZ administration were reported.
DISCUSSION
Despite these promising findings, randomized clinical trials to directly compare the efficacy and safety of TCZ and other currently available therapeutic options are needed.
PubMed: 38215463
DOI: 10.1097/IOP.0000000000002573 -
Frontiers in Endocrinology 2023There is emerging evidence which suggests the utility of artificial intelligence (AI) in the diagnostic assessment and pre-treatment evaluation of thyroid eye disease...
BACKGROUND
There is emerging evidence which suggests the utility of artificial intelligence (AI) in the diagnostic assessment and pre-treatment evaluation of thyroid eye disease (TED). This scoping review aims to (1) identify the extent of the available evidence (2) provide an in-depth analysis of AI research methodology of the studies included in the review (3) Identify knowledge gaps pertaining to research in this area.
METHODS
This review was performed according to the 2020 Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement (PRISMA). We quantify the diagnostic accuracy of AI models in the field of TED assessment and appraise the quality of these studies using the modified QUADAS-2 tool.
RESULTS
A total of 13 studies were included in this review. The most common AI models used in these studies are convolutional neural networks (CNN). The majority of the studies compared algorithm performance against healthcare professionals. The overall risk of bias and applicability using the modified Quality Assessment of Diagnostic Accuracy Studies 2 (QUADAS-2) tool led to most of the studies being classified as low risk, although higher deficiency was noted in the risk of bias in flow and timing.
CONCLUSIONS
While the results of the review showed high diagnostic accuracy of the AI models in identifying features of TED relevant to disease assessment, deficiencies in study design causing study bias and compromising study applicability were noted. Moving forward, limitations and challenges inherent to machine learning should be addressed with improved standardized guidance around study design, reporting, and legislative framework.
Topics: Humans; Algorithms; Artificial Intelligence; Graves Ophthalmopathy; Machine Learning; Neural Networks, Computer
PubMed: 38174334
DOI: 10.3389/fendo.2023.1300196 -
Ophthalmic Plastic and Reconstructive... Dec 2023MRI may potentially detect active thyroid eye disease prior to elevation of clinical activity score. We aimed to systematically review the existing literature pertaining...
PURPOSE
MRI may potentially detect active thyroid eye disease prior to elevation of clinical activity score. We aimed to systematically review the existing literature pertaining to MRI thyroid eye disease and to assess the role of MRI in the diagnosis of thyroid eye disease.
METHODS
A Population, Intervention, Comparison, Outcome/Preferred Reporting Items for Systematic Reviews and Meta-Analyses selection criteria was applied to identify studies for inclusion published between the years 2000 and 2023.
RESULTS
Twenty-four articles were identified for inclusion in the systematic review. All included studies utilized MRI as the imaging modality. MRI sequences used included T2-weighted imaging in 87.5%, T1-weighted imaging in 54.2%, diffusion-weighted imaging in 20.8%, and short tau inversion recovery in 16.7%. The most common parameters quantified were signal intensity ratio in 10 studies (41.7%) and T2-relaxation time in 8 studies (33.3%). Signal intensity ratio and T2-relaxation time were shown to correlate with clinical activity score and identify the phase of the disease.
CONCLUSIONS
MRI has untapped potential for further elucidating the highly complex biological processes in thyroid eye disease. As we move away from clinical activity score as a predictor of response to biologic therapy, MRI may prove more important than ever in the risk-benefit analysis around the use of immunomodulators.
Topics: Humans; Graves Ophthalmopathy; Magnetic Resonance Imaging
PubMed: 38054988
DOI: 10.1097/IOP.0000000000002511