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Plastic and Reconstructive Surgery.... Jun 2024Chronic lymphocytic leukemia is characterized by the accumulation of mature CD5-positive B-cells in the lymphoid organs. Extranodal involvement occurs in up to 10% of...
Chronic lymphocytic leukemia is characterized by the accumulation of mature CD5-positive B-cells in the lymphoid organs. Extranodal involvement occurs in up to 10% of cases and can arise in various tissues, including the orbit. Less than 400 cases of orbital lymphoma are diagnosed per year in the United States, typically manifesting as a form of B-cell non-Hodgkin lymphoma, with extranodal marginal zone B-cell lymphoma being the most common subtype. Orbital lymphoma typically presents with proptosis and a palpable mass; however, patients may also have a relatively benign examination. Here, we present a 76-year-old man with symmetric dermatochalasis and marked fat prolapse of all four lids, who was incidentally diagnosed with secondary orbital lymphoma in all four eyelids during a cosmetic four lid blepharoplasty. His history was significant for RAI Stage 0 chronic lymphocytic leukemia diagnosed 15 years before consultation. Orbital lymphoma presenting as orbital fat prolapse has only been reported a few times in the literature. To our knowledge, this is the first case of secondary orbital lymphoma in all four eyelids found incidentally during an aesthetic four lid blepharoplasty.
PubMed: 38855135
DOI: 10.1097/GOX.0000000000005870 -
Journal of Translational Medicine Jun 2024The pathogenesis of thyroid-associated orbitopathy (TAO) remains incompletely understand. The interaction between immunocytes and orbital fibroblasts (OFs) play a...
BACKGROUND
The pathogenesis of thyroid-associated orbitopathy (TAO) remains incompletely understand. The interaction between immunocytes and orbital fibroblasts (OFs) play a critical role in orbital inflammatory and fibrosis. Accumulating reports indicate that a significant portion of plasma exosomes (Pla-Exos) are derived from immune cells; however, their impact upon OFs function is unclear.
METHODS
OFs were primary cultured from inactive TAO patients. Exosomes isolated from plasma samples of patients with active TAO and healthy controls (HCs) were utilized for functional and RNA cargo analysis. Functional analysis in thymocyte differentiation antigen-1 (Thy-1) OFs measured expression of inflammatory and fibrotic markers (mRNAs and proteins) and cell activity in response to Pla-Exos. RNA cargo analysis was performed by RNA sequencing and RT-qPCR. Thy-1 OFs were transfected with miR-144-3p mimics/inhibitors to evaluate its regulation of inflammation, fibrosis, and proliferation.
RESULTS
Pla-Exos derived from active TAO patients (Pla-Exos) induced stronger production of inflammatory cytokines and hyaluronic acid (HA) in Thy-1 OFs while inhibiting their proliferation. Kyoto Encyclopedia of Genes and Genomes (KEGG) pathway analysis and single sample gene set enrichment analysis (ssGSEA) suggested that the difference in mRNA expression levels between Pla-Exos and Pla-Exos was closely related to immune cells. Differential expression analysis revealed that 62 upregulated and 45 downregulated miRNAs in Pla-Exos, with the elevation of miR-144-3p in both Pla-Exos and PBMCs in active TAO group. KEGG analysis revealed that the target genes of differentially expressed miRNA and miR-144-3p enriched in immune-related signaling pathways. Overexpression of the miR-144-3p mimic significantly upregulated the secretion of inflammatory cytokines and HA in Thy-1 OFs while inhibiting their proliferation.
CONCLUSION
Pla-Exos derived from patients with active TAO were immune-active, which may be a long-term stimulus casual for inflammatory and fibrotic progression of TAO. Our finding suggests that Pla-Exos could be used as biomarkers or treatment targets in TAO patients.
Topics: Humans; Exosomes; Graves Ophthalmopathy; Fibrosis; MicroRNAs; Fibroblasts; Orbit; Inflammation; Female; Male; Cell Proliferation; Middle Aged; Adult; Hyaluronic Acid; Cytokines; Thy-1 Antigens
PubMed: 38849907
DOI: 10.1186/s12967-024-05263-y -
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 -
Ultraschall in Der Medizin (Stuttgart,... Jun 2024Until now, ultrasound examination of the fetal eyes has not played an important role in prenatal diagnosis. National and international guidelines are generally confined...
Until now, ultrasound examination of the fetal eyes has not played an important role in prenatal diagnosis. National and international guidelines are generally confined to documentation of the presence of the orbits and the lenses. However, in recent years, with the advent of high-resolution ultrasound technology and increasing knowledge of prenatal medicine and genetics, careful examination of the fetal eye has enabled the detection of many ocular malformations before birth. This article provides an overview of the anatomy related to the development of the fetal eye and covers the following conditions: hypertelorism, hypotelorism, exophthalmos, microphthalmos, coloboma, cataract, persistent hyperplastic primary vitreous, retinal detachment, dacryocystocele, and septooptic dysplasia, etc. It is designed to illustrate the spectrum of ocular malformations and their appearance on prenatal ultrasound and to discuss their clinical impact and association with various syndromes.
PubMed: 38848751
DOI: 10.1055/a-2318-5464 -
European Thyroid Journal Jun 2024This study aims to report correlations between thyroid-stimulating immunoglobulin(TSI) and both clinical and radiological parameters in recent-onset symptomatic thyroid...
PURPOSE
This study aims to report correlations between thyroid-stimulating immunoglobulin(TSI) and both clinical and radiological parameters in recent-onset symptomatic thyroid eye disease(TED) patients.
METHODS
A prospective cohort study of TED patients managed at the Chinese University of Hong Kong from January 2014 to May 2022. Serum TSI levels were determined with the functional assay. Outcomes included the clinical activity score(CAS), marginal reflex distance1(MRD1), extraocular muscle motility restriction(EOMy), exophthalmos, and diplopia. The radiological assessment included cross-sectional areas and signal of extraocular muscles on STIR-sequence MRI.
RESULTS
A total of 255(197female) treatment-naïve patients, with an average onset age of 50±14 years, were included. Elevated pre-treatment TSI level was observed in 223(88%) patients. There was a weak positive correlation between TSI and CAS(r=0.28, P=0.000031), MRD1(r=0.17, P=0.0080), and the size of the levator palpebrae superioris/superior rectus complex(r=0.25, P=0.018). No significant correlation existed between TSI and STIR signals. The AUC and optimal cut-off value for clinical active TED were 0.67(95% confidence interval:0.60-0.75) and 284%(Specificity:50%, sensitivity:85%). 64 patients received intravenous methylprednisolone (IVMP) during the study interval, and they had a higher baseline TSI level than those who did not have IVMP(P=0.000044). Serial post-IVMP TSI among the 62 patients showed a significant reduction compared to the baseline level(P<0.001). Both the baseline and post-IVMP TSI levels, and percentages of TSI changes were comparable between patients who responded and non-responded to the first course of IVMP.
CONCLUSION
TSI can be a serum biomarker for the diagnosis, prognosis, and treatment response of TED. Further validation should be further warranted.
PubMed: 38847819
DOI: 10.1530/ETJ-23-0129 -
European Journal of Ophthalmology Jun 2024Posterior movement of ocular tissue secondary to orbital cavernous venous malformation shrinkage from fractionated stereotactic radiotherapy can allow healthy structures...
BACKGROUND
Posterior movement of ocular tissue secondary to orbital cavernous venous malformation shrinkage from fractionated stereotactic radiotherapy can allow healthy structures to move into the radiation field during treatment. This may carry an increased risk of radiation-induced retinopathy.
METHODS
We present a case of a young female whose radiotherapy treatment for an orbital cavernous venous malformation resulted in a 3 mm reduction in proptosis and subsequent retinopathy.
RESULTS
The severity of the patient's radiation-induced radiotherapy exceeded expectations. The venous malformation shrinkage during treatment and ensuing posterior movement of the globe suggested an increased involvement of ocular tissue in the radiation field, prompting consideration of interval neuroimaging and tumour mapping.
CONCLUSIONS
We describe and suggest a protocol of onboard neuroimaging during the radiation therapy course to better target tumour volumes and minimise collateral tissue damage. To our knowledge, this has not been previously described in the ophthalmic literature.
PubMed: 38847131
DOI: 10.1177/11206721241259797 -
Annals of Medicine and Surgery (2012) Jun 2024Carotid cavernous fistulas are uncommon vascular abnormalities marked by anomalous connections between the carotid artery and the cavernous sinus. The authors present a...
INTRODUCTION
Carotid cavernous fistulas are uncommon vascular abnormalities marked by anomalous connections between the carotid artery and the cavernous sinus. The authors present a case of a direct carotid cavernous fistula and its successful treatment in a 42-year-old female.
CASE PRESENTATION
A 42-year-old female presented with right eye painful swelling and visual disturbance. She had no known comorbidities or history of injury. Examination showed proptosis, chemosis, and orbital bruit. Carotid angiography confirmed a carotid cavernous fistula, which was managed endovascularly. The patient fully recovered after treatment.
DISCUSSION
Carotid cavernous fistula occurs spontaneously or as a result of trauma or other vascular abnormalities. Common clinical manifestations include proptosis, chemosis, and orbital bruit, with vision loss being a feared complication. Diagnosis is typically confirmed through angiography, with digital subtraction angiography being the gold standard. Endovascular treatment is usually effective, although surgical management may be necessary in certain cases.
CONCLUSION
Carotid cavernous fistula is a rare but potentially sight-threatening neurological condition. Treatment with a transvenous approach is effective for the management of direct carotid cavernous fistula.
PubMed: 38846860
DOI: 10.1097/MS9.0000000000002151 -
Cureus May 2024Optic nerve schwannoma is a very rarely occurring tumor described in the literature. It is due to the fact that the optic nerve is myelinated by oligodendrocytes....
Optic nerve schwannoma is a very rarely occurring tumor described in the literature. It is due to the fact that the optic nerve is myelinated by oligodendrocytes. Schwannomas are tumors of the peripheral nervous system, hence optic nerve schwannoma is a rare phenomenon. A 34-year-old patient presented in the outpatient department with complaints of gradual painless protrusion of the left eye (LE) for the past one year. There was no history of diminution of vision. On examination, vision in both eyes was 6/6, anterior segment examination in both eyes was normal, and pupils were central, circular, and reacting to light. Intraocular pressure was measured on a noncontact tonometer and was within normal range. Both eyes' optic disc, fundus, and visual fields were normal. On inspection, axial proptosis was noted in the LE. Proptosis measurement (on Hertel exophthalmometer) in the right eye was 17 mm and in the left eye was 21 mm. MRI of the orbit without contrast was done and showed a well-defined, soft tissue lesion of the optic nerve in the intraconal compartment of the left orbit. Surgical excision of the tumor was done by lateral orbitotomy approach and the tumor was removed in total. Histopathological examination of the mass revealed a benign spindle cell neoplasm suggestive of schwannoma. Postoperatively, proptosis was resolved, 17 mm both in the right and left eye (on Hertel exophthalmometer), and vision in LE remained unchanged (6/6). Postoperatively, intraocular pressure (on noncontact tonometer) was within normal range, and the optic disc, fundus, and visual fields were normal.
PubMed: 38846181
DOI: 10.7759/cureus.59824 -
The Journal of Craniofacial Surgery Jun 2024Thyroid eye disease (TED) is characterized by a variety of disfiguring periocular changes. Vertical globe changes affecting the relative position of the eyelids are not...
PURPOSE
Thyroid eye disease (TED) is characterized by a variety of disfiguring periocular changes. Vertical globe changes affecting the relative position of the eyelids are not well understood in patients with TED. This study seeks to determine the effect of orbital decompression on vertical globe displacement in patients with TED, without TED, and with intraconal tumor (ICT).
METHODS
For this cross-sectional study, a clinical database was used to identify patients with TED. Comparison groups were drawn from separate anonymized databases. Vertical position and interpupillary distance (IPD) were measured from photographs and exophthalmos was measured via Hertel's exophthalmometer. Primary outcomes were vertical globe position at baseline and postoperatively in patients with TED and ICT. Secondary outcomes included the relationship between vertical globe position, exophthalmos, and IPD.
RESULTS
Among 269 participants meeting the inclusion criteria, mean vertical globe position was significantly lower in patients with TED following lateral decompression surgery compared to controls, after accounting for race, age, and sex. While patients with ICT had a significant difference in preoperative and postoperative IPD, patients with TED did not. Medial or inferior decompression did not significantly change globe position and lateral decompression did not cause lateral canthal dystopia in patients with TED. No association between postoperative changes in exophthalmometry, IPD, and globe position was found in patients with TED.
CONCLUSIONS
Patients with TED experience hypoglobus that does not improve following decompression surgery. There was no correlation between change in vertical globe position and exophthalmos or IPD among patients with TED. Surgeons should discuss the possibility of hypoglobus as a persistent finding for patients with TED undergoing decompression surgery.
PubMed: 38838355
DOI: 10.1097/SCS.0000000000010345 -
Endocrine Reviews Jun 2024Thyroid eye disease (TED) is the most common extra thyroidal manifestation of Graves' disease (GD). It may also present in those who are hypothyroid or euthyroid. The...
Thyroid eye disease (TED) is the most common extra thyroidal manifestation of Graves' disease (GD). It may also present in those who are hypothyroid or euthyroid. The characteristic clinical manifestations of TED: chemosis, lid swelling, proptosis and diplopia are driven by a combination of inflammation and extracellular matrix modification. It has recently emerged that one of the major drivers of this molecular signature is the over-expression of the insulin like growth factor-1 receptor (IGF-1R) on key effector cells in TED pathogenesis. The overexpression of the IGF-1R is coupled with a dysregulation of the IGF-1R axis, which links other pathways that modulate inflammation, such as fibrosis and extracellular matrix organization, in patients with TED. This overexpression is also found to persist from the acute stage into the chronic phase. Teprotumumab, a fully human immunoglobulin (Ig) G1 monoclonal antibody that inhibits the IGF-1R, recently gained approval in the US for the treatment of TED. In phase 2 and phase 3 clinical studies, teprotumumab showed efficacy in reducing inflammation, proptosis, diplopia and burden on quality of life, in patients who were treated. Post introduction studies have confirmed the results of the phase 2 and phase 3 studies. Since 2020, over 5, 800 patients have been treated with teprotumumab and it appears to be well tolerated. The American Thyroid Association and the European Thyroid Association have recommended it as first line therapy for patients with moderate to severe TED, who display features of proptosis and diplopia.
PubMed: 38838219
DOI: 10.1210/endrev/bnae018