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2.
Int Ophthalmol ; 44(1): 206, 2024 Apr 27.
Article in English | MEDLINE | ID: mdl-38678129

ABSTRACT

PURPOSE: Patients with thyroid eye disease (TED) can present with asymmetric disease. The purpose of this study was to evaluate the prevalence of asymmetric TED in an Australian cohort and investigate clinical, biochemical, and radiological associations of asymmetric TED. METHODS: This was a retrospective study of patients with TED who underwent Hertel exophthalmometry and orbital computed tomography (CT) scans. Asymmetry was defined as a difference in the globe protrusion of ≥ 3 mm using Hertel exophthalmometry. Data was collected on patient demographics, clinical disease presentation, thyroid function tests and antibody levels. Muscles volumes were determined by manually segmenting the extraocular muscles on CT scans using a commercially available software. RESULTS: 172 orbits from 86 patients were included in the study. 28 (33%) patients had asymmetric TED. No significant differences were observed in age, gender, thyroid hormone status, anti-thyroid peroxidase levels, thyroid stimulating hormone receptor antibodies, disease activity status, or dysthyroid optic neuropathy between the asymmetric and non-asymmetric groups. The extraocular muscle volumes were significantly higher in the asymmetric orbit compared to its contralateral orbit. CONCLUSION: Asymmetric TED is a reasonably common occurrence in the course of TED. It is associated with higher extraocular muscle volumes, suggesting muscle enlargement as one of the underlying contributors to asymmetric proptosis. Thyroid eye disease should be considered in the differential of asymmetric orbital inflammatory disease.


Subject(s)
Exophthalmos , Graves Ophthalmopathy , Oculomotor Muscles , Tomography, X-Ray Computed , Humans , Male , Female , Retrospective Studies , Exophthalmos/diagnosis , Graves Ophthalmopathy/diagnosis , Graves Ophthalmopathy/complications , Middle Aged , Oculomotor Muscles/physiopathology , Oculomotor Muscles/diagnostic imaging , Oculomotor Muscles/pathology , Aged , Adult , Orbit/diagnostic imaging , Prevalence , Aged, 80 and over , Australia/epidemiology
3.
Acta Neurochir (Wien) ; 166(1): 164, 2024 Apr 02.
Article in English | MEDLINE | ID: mdl-38564027

ABSTRACT

OBJECTIVE: This study aimed to identify factors affecting proptosis recovery in spheno-orbital meningioma (SOM) surgery and assess functional and oncological outcomes. METHODS: Data from 32 consecutive SOM surgery cases (2002-2021) were analyzed. Clinical, radiological, operative, and oncological parameters were examined. Proptosis was assessed using the exophthalmos index (EI) on MRI or CT scans. Statistical analyses were performed to identify predictive factors for proptosis recovery. RESULTS: Proptosis improved in 75% of patients post-surgery (EI decreased from 1.28 ± 0.16 to 1.20 ± 0.13, p = 0.048). Patients with stable or worsened EI had higher body mass index (28.5 ± 7.9 vs. 24.1 ± 4.7, p = 0.18), Simpson grade (IV 75% vs. 65%, p = 0.24), and middle sphenoid wing epicenter involvement (63% vs. 38%, p = 0.12), but no significant factors were associated with unfavorable exophthalmos outcomes. The improvement group had higher en plaque morphology, infratemporal fossa invasion, and radiation treatment for cavernous sinus residual tumor (88% vs. 75%, p = 0.25; 51% vs. 25%, p = 0.42; 41% vs. 25%, p = 0.42, respectively), but without statistical significance. Visual acuity remained stable in 78%, improved in 13%, and worsened in 9% during follow-up. Surgery had a positive impact on preoperative oculomotor nerve dysfunction in 3 of 4 patients (75%). Postoperative oculomotor nerve dysfunction was observed in 25%, of which 75% fully recovered. This occurrence was significantly associated with irradiation of an orbital tumor residue (p = 0.04). New postoperative trigeminal hypoesthesia was observed in 47%, of which 73% recovered. All SOMs were classified as WHO grade 1, and complementary treatments achieved oncological control, requiring gamma-knife radiosurgery in 53% and standard radiotherapy in 6%. CONCLUSIONS: Surgery effectively improves proptosis in SOM, though complete resolution is rare. The absence of predictive factors suggests multifactorial causes, including body mass index and tumor resection grade. Postoperative oculomotor nerve dysfunction and trigeminal hypoesthesia are common but often recover. Gamma-knife radiosurgery maintains long-term oncological control for evolving tumor residue.


Subject(s)
Exophthalmos , Meningeal Neoplasms , Meningioma , Humans , Meningioma/complications , Meningioma/diagnostic imaging , Meningioma/surgery , Hypesthesia , Exophthalmos/etiology , Exophthalmos/surgery , Body Mass Index , Meningeal Neoplasms/complications , Meningeal Neoplasms/diagnostic imaging , Meningeal Neoplasms/surgery
4.
Sci Rep ; 14(1): 9497, 2024 04 25.
Article in English | MEDLINE | ID: mdl-38664418

ABSTRACT

Raine syndrome (RNS) is a rare autosomal recessive osteosclerotic dysplasia. RNS is caused by loss-of-function disease-causative variants of the FAM20C gene that encodes a kinase that phosphorylates most of the secreted proteins found in the body fluids and extracellular matrix. The most common RNS clinical features are generalized osteosclerosis, facial dysmorphism, intracerebral calcifications and respiratory defects. In non-lethal RNS forms, oral traits include a well-studied hypoplastic amelogenesis imperfecta (AI) and a much less characterized gingival phenotype. We used immunomorphological, biochemical, and siRNA approaches to analyze gingival tissues and primary cultures of gingival fibroblasts of two unrelated, previously reported RNS patients. We showed that fibrosis, pathological gingival calcifications and increased expression of various profibrotic and pro-osteogenic proteins such as POSTN, SPARC and VIM were common findings. Proteomic analysis of differentially expressed proteins demonstrated that proteins involved in extracellular matrix (ECM) regulation and related to the TGFß/SMAD signaling pathway were increased. Functional analyses confirmed the upregulation of TGFß/SMAD signaling and subsequently uncovered the involvement of two closely related transcription cofactors important in fibrogenesis, Yes-associated protein (YAP) and transcriptional coactivator with PDZ-binding motif (TAZ). Knocking down of FAM20C confirmed the TGFß-YAP/TAZ interplay indicating that a profibrotic loop enabled gingival fibrosis in RNS patients. In summary, our in vivo and in vitro data provide a detailed description of the RNS gingival phenotype. They show that gingival fibrosis and calcifications are associated with, and most likely caused by excessed ECM production and disorganization. They furthermore uncover the contribution of increased TGFß-YAP/TAZ signaling in the pathogenesis of the gingival fibrosis.


Subject(s)
Abnormalities, Multiple , Adaptor Proteins, Signal Transducing , Cleft Palate , Dental Enamel Hypoplasia , Exophthalmos , Fibroblasts , Fibrosis , Gingiva , Osteosclerosis , Proteomics , Signal Transduction , Transcription Factors , Transforming Growth Factor beta , YAP-Signaling Proteins , Humans , Transforming Growth Factor beta/metabolism , Gingiva/metabolism , Gingiva/pathology , Proteomics/methods , Fibrosis/metabolism , YAP-Signaling Proteins/metabolism , YAP-Signaling Proteins/genetics , Osteosclerosis/metabolism , Osteosclerosis/genetics , Osteosclerosis/pathology , Adaptor Proteins, Signal Transducing/metabolism , Adaptor Proteins, Signal Transducing/genetics , Transcription Factors/metabolism , Transcription Factors/genetics , Dental Enamel Hypoplasia/metabolism , Dental Enamel Hypoplasia/genetics , Dental Enamel Hypoplasia/pathology , Fibroblasts/metabolism , Fibroblasts/pathology , Microcephaly/metabolism , Microcephaly/genetics , Microcephaly/pathology , Female , Transcriptional Coactivator with PDZ-Binding Motif Proteins/metabolism , Male , Trans-Activators/metabolism , Trans-Activators/genetics , Intracellular Signaling Peptides and Proteins/metabolism , Intracellular Signaling Peptides and Proteins/genetics , Casein Kinase I/metabolism , Casein Kinase I/genetics , Extracellular Matrix Proteins/metabolism , Extracellular Matrix Proteins/genetics , Amelogenesis Imperfecta/metabolism , Amelogenesis Imperfecta/genetics , Amelogenesis Imperfecta/pathology , Cells, Cultured
5.
J Med Case Rep ; 18(1): 164, 2024 Apr 07.
Article in English | MEDLINE | ID: mdl-38582878

ABSTRACT

BACKGROUND: Graves' disease is the autoimmune activation of the thyroid gland causing diffuse enlargement and hyperfunction of the gland. Manifestations of Graves' disease are multisystemic and include thyroid orbitopathy; pretibial myxedema, also referred to as thyroid dermopathy; and thyroid acropachy, described as a severe form of thyroid dermopathy. Our paper focuses on an atypical case of thyroid dermopathy. CASE PRESENTATION: An 11-year-old Saudi male presented with a prominent diffuse goiter and exophthalmos. Investigations were consistent with a diagnosis of Graves' disease. The physical exam showed diffuse, non-pitting swelling of the ankle and penis, mimicking a lymphatic malformation. Further, multiple nodules were found on the hands and feet. Treatment of the nodules with cautery resulted in more severe nodules. CONCLUSION: This report describes rare presentations of thyroid dermopathy mimicking lymphatic malformation. The Koebner phenomenon can explain this patient's atypical presentations. Intralesional injections of triamcinolone and total thyroidectomy showed clear improvement.


Subject(s)
Exophthalmos , Graves Disease , Myxedema , Skin Diseases , Humans , Male , Child , Graves Disease/complications , Graves Disease/diagnosis , Myxedema/diagnosis , Myxedema/etiology
6.
Int Ophthalmol ; 44(1): 179, 2024 Apr 16.
Article in English | MEDLINE | ID: mdl-38622479

ABSTRACT

PURPOSE: To evaluate the clinical outcomes of intravenous tocilizumab (TCZ) injection in patients with moderate-to-severe active thyroid eye disease (TED). METHODS: Patients with active and moderate-to-severe TED who did not respond to conventional therapies were treated with TCZ from June 2019 to January 2021. The medical records of the patients were evaluated before the treatment. We analyzed patient demographics, including the duration of Graves' disease and TED, and assessed subjective symptoms, such as diplopia and ocular movement, clinical activity score (CAS), modified NOSPECS score, and exophthalmos before treatment and at 4, 8, 12, and 16 weeks after the first drug injection. Blood tests, including TSH Rc Ab and TS Ab, were performed before treatment and 24 weeks after the first injection. And orbital computed tomography (CT) was performed and Barrett's Index was calculated at baseline and after completion of all injections. RESULTS: Nineteen completed the scheduled treatment. There were no significant side effects, other than herpes zoster in one case and headache and dermatitis in another. Clinical symptoms before and 16 weeks after the treatment showed mean CAS decreased by 2.4 points, mean modified NOSPECS score decreased by 3.7 points, and mean exophthalmos decreased by 0.4 mm. Diplopia and extraocular muscle limitation improved in ten and remained stationary in five of the 15 patients, who presented with extraocular motility abnormalities. Six of 11 patients who underwent orbit CT showed improvement in muscle size. The mean TSH Rc Ab decreased by 7.5 IU/L and TS-Ab decreased by 162.9%. CONCLUSION: TCZ can treat active moderate-to-severe TED, showing high drug compliance and reasonable response to inflammation and extraocular motility abnormality.


Subject(s)
Antibodies, Monoclonal, Humanized , Exophthalmos , Graves Ophthalmopathy , Humans , Graves Ophthalmopathy/diagnosis , Graves Ophthalmopathy/drug therapy , Prospective Studies , Diplopia/etiology , Adrenal Cortex Hormones/therapeutic use , Thyrotropin
7.
Arch. Soc. Esp. Oftalmol ; 99(4): 173-176, abr. 2024. ilus
Article in Spanish | IBECS | ID: ibc-232139

ABSTRACT

Este manuscrito presenta un caso de un leiomioma orbitario de larga evolución en un joven de 14años. El tumor era inusualmente grande y causaba una proptosis severa y una afectación importante de la musculatura ocular. El paciente presentaba amaurosis, una oftalmoplejía completa, dolor ocular espontáneo e incapacidad para cerrar los párpados. Debido al tamaño del tumor y a su progresión, se realizó una exenteración orbitaria derecha para eliminar todo el contenido orbitario, incluyendo el tumor y el globo ocular. El procedimiento quirúrgico tenía como objetivo prevenir la recurrencia del tumor y mejorar la calidad de vida del paciente. El análisis histopatológico confirmó el diagnóstico de leiomioma orbitario. Este caso presenta un interés particular por el grado de evolución que ha alcanzado. La extirpación completa del tumor y un seguimiento a largo plazo son necesarios para prevenir la recurrencia y garantizar resultados óptimos para el paciente. Además, este caso refleja las grandes diferencias en el acceso a la sanidad en las diferentes regiones del mundo. (AU)


This manuscript describes an exceptional case of a long-standing orbital leiomyoma in a 14-year-old male. The tumor was unusually large, causing severe proptosis and significant involvement of the ocular muscles. The patient presented with amaurosis, complete ophthalmoplegia, spontaneous eye pain, and the inability to close the eyelids, leading to psychological distress. Due to the tumor's size and progression, a right orbital exenteration was performed to remove all orbital contents, including the tumor and the eyeball. The surgical procedure aimed to prevent tumor recurrence and improve the patient's quality of life. The histopathological analysis confirmed the diagnosis of orbital leiomyoma. This case presents a particular interest due to the degree of evolution it has reached. Complete tumor excision and long-term follow-up are necessary to prevent recurrence and ensure optimal patient outcomes. This report underscores global healthcare disparities and the complexity of managing rare orbital neoplasms in diverse country settings. (AU)


Subject(s)
Humans , Male , Adolescent , Leiomyoma , Exophthalmos , Ophthalmoplegia , Orbit Evisceration
10.
Int Ophthalmol ; 44(1): 147, 2024 Mar 19.
Article in English | MEDLINE | ID: mdl-38499845

ABSTRACT

BACKGROUND: This study aims to compare the changes in ophthalmic parameters among syndromic craniosynostosis patients who underwent craniofacial skeletal expansion procedures via distraction osteogenesis (DO). METHOD: A retrospective study was conducted involving syndromic craniosynostosis patients who underwent surgical expansion via the DO technique from the year 2012 to March 2022. Changes in six parameters which consist of visual acuity, refractive error, optic disc health, intraocular pressure, degree of proptosis and orbital volume were measured objectively pre and post-surgery. For categorical parameters, the Chi-square cross-tab test was done. Paired sample T-test was used for normally distributed variables. Wilcoxon signed-rank test was used for non-normally distributed data. RESULTS: Visual impairment was present in 21.4% of eyes before surgery and increased to 28.5% post-surgery. Three patients had changes of refractive error post-surgery with one developed hypermetropia, another developed anisometropia and the last had improvement to no refractive error. Two patients had optic disc swelling which was resolved post-surgery. Intraocular pressure changes were inconsistent post-surgery. All patients achieved a significant reduction in the degree of proptosis post-surgery. Orbital volume calculation using computed tomography (CT) scans shows a significant increase in volume post-surgery for all patients. CONCLUSION: Our study shows a significant increase in orbital volume post-surgery with a reduction in the degree of proptosis. Optic disc and nerve health improved after the surgery. Changes in terms of visual acuity, refractive error and IOP were inconsistent after the surgical intervention.


Subject(s)
Craniosynostoses , Exophthalmos , Osteogenesis, Distraction , Refractive Errors , Humans , Osteogenesis, Distraction/methods , Retrospective Studies , Craniosynostoses/diagnosis , Craniosynostoses/surgery , Refractive Errors/diagnosis
11.
Ophthalmic Plast Reconstr Surg ; 40(2): e41-e42, 2024.
Article in English | MEDLINE | ID: mdl-38427840

ABSTRACT

Superior ophthalmic vein thrombosis is a rare condition scarcely described in clinical literature with potentially severe consequences including permanent vision loss. This report details the case of a 70-year-old woman who presented with acute binocular horizontal diplopia, relative proptosis of the OD by 4 mm, and pain OD. On exam, visual acuity was 20/20 OD and 20/30 OS with full extraocular movements. CT revealed proptosis OD with a thrombosed superior ophthalmic varix. Evaluation for etiology of hypercoagulability was unremarkable, although the patient did have an asymptomatic COVID-19 infection 1 month prior. To the authors' knowledge, this is the first reported case of superior ophthalmic vein thrombosis secondary to an asymptomatic COVID-19 infection.


Subject(s)
COVID-19 , Exophthalmos , Thrombosis , Varicose Veins , Venous Thrombosis , Female , Humans , Aged , Venous Thrombosis/etiology , Venous Thrombosis/complications , COVID-19/complications , Eye/blood supply , Exophthalmos/etiology , Diplopia/etiology , Varicose Veins/complications
12.
Arq Bras Oftalmol ; 87(2): e20220241, 2024.
Article in English | MEDLINE | ID: mdl-38451680

ABSTRACT

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.


Subject(s)
Exophthalmos , Eye Diseases , Nasopharyngeal Carcinoma , Nasopharyngeal Neoplasms , Humans , Databases, Factual , Exophthalmos/etiology , Eye , Nasopharyngeal Carcinoma/complications , Nasopharyngeal Neoplasms/complications , Eye Diseases/etiology
13.
J Med Case Rep ; 18(1): 92, 2024 Mar 08.
Article in English | MEDLINE | ID: mdl-38454521

ABSTRACT

BACKGROUND: Orbital ischemic syndrome is a rare entity. The clinical signs typically demonstrate acute loss of visual acuity, chemosis, proptosis, ptosis, and total ophthalmoparesis. We report a case of a man who suffered an acute internal carotid artery occlusion and developed orbital ischemic syndrome after a mechanical thrombectomy. CASE PRESENTATION: A 57-year-old Vietnamese (Kinh ethnicity) man was brought to the emergency room with complaints of a speech disturbance, facial palsy, and severe weakness of the left arm and leg, which had started 4 hours earlier, after waking up. The National Institutes of Health Stroke Scale 12 (NIHSS 12) revealed the neurological score at admission. A head computed tomography scan showed no intracranial bleeding and an Alberta Stroke Program Early Computed Tomography Score (ASPECTS) of 8 on the right brain. Computed tomography angiography showed an occlusion of the right internal carotid artery. After that, a mechanical thrombectomy was performed, and the internal carotid artery was completely reperfused. After 10 hours, he experienced orbital pain, proptosis, ptosis, chemosis, and ophthalmoplegia of the right orbit. He also had acute loss of visual acuity, and fundoscopic examination revealed papilledema, no retinal hemorrhage, and no bruit in orbital auscultation. Intraocular pressure in the right eye was measured at 50.5 mmHg. Computed tomography angiography showed no carotid-cavernous fistula, but slight enlargement of the right medial and lateral rectus muscles. He was treated with steroids and hyperosmolar agents and recovered 7 days later, but had persistent loss of visual acuity in the right eye. CONCLUSION: Orbital ischemic syndrome is a rare complication after mechanical thrombectomy treatment in acute ischemic stroke that can lead to loss of visual acuity.


Subject(s)
Exophthalmos , Ischemic Stroke , Ophthalmoplegia , Stroke , Humans , Male , Middle Aged , Carotid Artery, Internal/diagnostic imaging , Infarction/complications , Ischemia , Ophthalmoplegia/etiology , Stroke/complications , Thrombectomy/adverse effects , Thrombectomy/methods
14.
J Craniomaxillofac Surg ; 52(4): 432-437, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38448333

ABSTRACT

This study aimed to assess the feasibility of utilizing a surgical guide, designed through digital medical technology, in lateral orbital decompression surgery. METHODS: In total, 18 patients with thyroid-associated ophthalmopathy (TAO), who underwent orbital balance decompression surgery at the Affiliated Eye Hospital of Nanchang University between September 2018 and August 2022, were included. Orbital CT scanning was performed on all patients with TAO, and Mimics 21.0 software was used to reconstruct a three-dimensional model of the orbit based on the CT data. The osteotomy guide plate for lateral orbital decompression surgery was designed using 3-matic 13.0 software, adhering to the criteria of surgical effectiveness and safety. The surgical positioning guide was designed using Geomagic Wrap 21.0. Once printed, the surgical guide was sterilized with low-temperature plasma and applied during surgery. Of the nine patients treated using a surgical navigation system, three cases experienced cerebrospinal fluid leakage complications during the procedure, and two exhibited inadequate bone removal along the lateral wall. In contrast, among the nine patients treated with surgical guides, no intraoperative cerebrospinal fluid leakage or evidence of insufficient lateral wall bone removal was observed, highlighting a statistically significant distinction between the two cohorts (p = 0.046). Postoperative improvements were notable in best-corrected visual acuity (BCVA) and exophthalmos for patients afflicted with extremely severe TAO. The surgical guide, designed with digital medical technology, has been shown to be an effective and secure auxiliary tool in lateral orbital decompression surgery. It not only aids in reducing the incidence of intraoperative complications, but also enhances the accuracy and safety of surgery. These improvements offer robust support for continued exploration in this field within clinical practice.


Subject(s)
Exophthalmos , Graves Ophthalmopathy , Humans , Orbit/surgery , Decompression, Surgical/methods , Retrospective Studies , Graves Ophthalmopathy/surgery , Exophthalmos/surgery , Cerebrospinal Fluid Leak/surgery
15.
Endocr Pract ; 30(5): 470-475, 2024 May.
Article in English | MEDLINE | ID: mdl-38341128

ABSTRACT

OBJECTIVE: In thyroid eye disease (TED), inflammation and expansion of orbital muscle and periorbital fat result in diplopia and proptosis, severely impacting patient quality of life (QOL). The reported health state utility (HSU) scores, which are QOL measures, allow quantification of TED impact and improvement with therapies; however, no current QOL instrument has been validated with HSU scores for TED. Here, we used the disease-specific Graves Ophthalmopathy Quality of Life (GO-QOL) questionnaire and HSU scores to validate QOL impact. METHODS: The GO-QOL scores from patients in 2 randomized, masked, placebo-controlled teprotumumab trials (N=171) were compared with 6 HSU values based on severity of proptosis/diplopia in those studies. Patient GO-QOL and HSU scores were compared at baseline and after 6-month treatment via regression analyses. GO-QOL and HSU scores were correlated for validation and quantification of QOL impact by severity state and to estimate quality-adjusted life year improvement. RESULTS: GO-QOL scores were correlated with TED severity, indicating that worse severity was associated with lower (worse) GO-QOL scores. Less severe health states were represented by higher (better) GO-QOL scores. Importantly, GO-QOL scores were positively correlated with utility scores of the 6 health states, allowing for conversion of the GO-QOL scores to utility scores. A positive (improved) 0.013 utility change was found for each 1-point (positive) improvement in GO-QOL score produced by teprotumumab versus placebo. CONCLUSION: Patients with moderate-to-severe active TED health states demonstrate increasing TED severity associated with declining utility values and worsening GO-QOL scores. These results indicate that the GO-QOL scores can be used to bridge to the HSU scores for benefit quantification.


Subject(s)
Antibodies, Monoclonal, Humanized , Graves Ophthalmopathy , Quality of Life , Adult , Aged , Female , Humans , Male , Middle Aged , Antibodies, Monoclonal, Humanized/therapeutic use , Exophthalmos , Graves Ophthalmopathy/psychology , Graves Ophthalmopathy/drug therapy , Health Status , Severity of Illness Index , Surveys and Questionnaires
16.
Oral Maxillofac Surg ; 28(2): 999-1004, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38378934

ABSTRACT

PURPOSE: We report a case of fibrous dysplasia (FD) with aneurysmal bone cyst (ABC)-like change in a child with orbital involvement, review the related cases, and discuss clinical features, therapy, and prognosis of this disease. CASE PRESENTATION: A 10-year-old girl had right proptosis (degree of exophthalmos: OD 16 mm, OS 13 mm) and limited vision (visual acuity: OD 1.0, OS 0.8) without trauma. Preoperative CT showed a 5.0*4.3 cm right-sided crania-orbital communicating tumor. MRI indicated a well-defined multicystic mass with scattered fluid levels and soap bubble-like alterations. The child underwent total tumor resection and orbital parietal titanium mesh reconstruction. At 20 months of follow-up, the child has recovered from ocular problems, and the tumor has not recurred. CONCLUSION: FD combined with ABC rarely occurs in orbit and generally begins with ocular symptoms. The etiology is uncertain. Early diagnosis and surgery are essential. Complete resection is suggested whenever possible because residual lesions may recur.


Subject(s)
Bone Cysts, Aneurysmal , Humans , Female , Child , Bone Cysts, Aneurysmal/surgery , Bone Cysts, Aneurysmal/diagnostic imaging , Bone Cysts, Aneurysmal/pathology , Exophthalmos/etiology , Exophthalmos/surgery , Tomography, X-Ray Computed , Orbital Diseases/surgery , Orbital Diseases/diagnostic imaging , Orbital Diseases/pathology , Magnetic Resonance Imaging , Fibrous Dysplasia of Bone/surgery , Fibrous Dysplasia of Bone/diagnostic imaging , Fibrous Dysplasia of Bone/complications , Surgical Mesh , Plastic Surgery Procedures/methods
17.
PLoS Pathog ; 20(2): e1012033, 2024 Feb.
Article in English | MEDLINE | ID: mdl-38421944

ABSTRACT

The host environment is of critical importance for antibiotic efficacy. By impacting bacterial machineries, stresses encountered by pathogens during infection promote the formation of phenotypic variants that are transiently insensitive to the action of antibiotics. It is assumed that these recalcitrant bacteria-termed persisters-contribute to antibiotic treatment failure and relapsing infections. Recently, we demonstrated that host reactive nitrogen species (RNS) transiently protect persisters against the action of ß-lactam antibiotics by delaying their regrowth within host cells. Here, we discovered that RNS intoxication of persisters also collaterally sensitizing them to fluoroquinolones during infection, explaining the higher efficiency of fluoroquinolones against intramacrophage Salmonella. By reducing bacterial respiration and the proton-motive force, RNS inactivate the AcrAB efflux machinery of persisters, facilitating the accumulation of fluoroquinolones intracellularly. Our work shows that target inactivity is not the sole reason for Salmonella persisters to withstand antibiotics during infection, with active efflux being a major contributor to survival. Thus, understanding how the host environment impacts persister physiology is critical to optimize antibiotics efficacy during infection.


Subject(s)
Abnormalities, Multiple , Anti-Bacterial Agents , Cleft Palate , Exophthalmos , Fluoroquinolones , Microcephaly , Osteosclerosis , Anti-Bacterial Agents/pharmacology , Biological Transport , Monobactams , Proton-Motive Force
18.
Arch Soc Esp Oftalmol (Engl Ed) ; 99(4): 173-176, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38309659

ABSTRACT

This manuscript describes an exceptional case of a long-standing orbital leiomyoma in a 14-year-old male. The tumor was unusually large, causing severe proptosis and significant involvement of the ocular muscles. The patient presented with amaurosis, complete ophthalmoplegia, spontaneous eye pain, and the inability to close the eyelids, leading to psychological distress. Due to the tumor's size and progression, a right orbital exenteration was performed to remove all orbital contents, including the tumor and the eyeball. The surgical procedure aimed to prevent tumor recurrence and improve the patient's quality of life. The histopathological analysis confirmed the diagnosis of orbital leiomyoma. This case presents a particular interest due to the degree of evolution it has reached. Complete tumor excision and long-term follow-up are necessary to prevent recurrence and ensure optimal patient outcomes. This report underscores global healthcare disparities and the complexity of managing rare orbital neoplasms in diverse country settings.


Subject(s)
Exophthalmos , Leiomyoma , Muscle Neoplasms , Orbital Neoplasms , Male , Humans , Child , Adolescent , Orbital Neoplasms/diagnostic imaging , Orbital Neoplasms/surgery , Quality of Life , Neoplasm Recurrence, Local , Exophthalmos/etiology , Muscle Neoplasms/complications , Leiomyoma/diagnostic imaging , Leiomyoma/surgery , Leiomyoma/complications
19.
Int Ophthalmol ; 44(1): 81, 2024 Feb 15.
Article in English | MEDLINE | ID: mdl-38358400

ABSTRACT

BACKGROUND: The main treatment for the symptoms of proptosis and optic nerve compression caused by thyroid-associated ophthalmopathy is orbital decompression surgery. Medial inferior wall decompression and balanced decompression are two frequently used surgical procedures. However, there is no unified consensus on how to choose different surgical options for orbital decompression in clinical practice. AIMS: To compare the effects of medial inferior wall decompression and balanced decompression surgery through meta-analysis and to provide reference for clinical optimal decision making. METHODS: Databases, including PubMed, Web of Science, Ovid, Cochrane Library, and ClinicalTrials.gov, were searched for randomized controlled trials and cohort studies on decompression surgery for thyroid-associated ophthalmopathy published from inception to March 21, 2023. Using RevMan 5.3 software, a meta-analysis was conducted based on the following outcome indicators: proptosis, diplopia rate, intraocular pressure, visual acuity, and complication rate. RESULTS: Two randomized controlled trials and five cohort studies with a total of 377 patients were included in this analysis. After balanced decompression surgery, patients with thyroid-associated ophthalmopathy experienced a significant decrease in proptosis [MD = 4.92, 95% CI (4.26, 5.58), P < 0.0001]. Balanced decompression can improve postoperative visual acuity [MD = - 0.35, 95% CI (- 0.56, - 0.13), P = 0.001] and intraocular pressure [MD = 5.33, 95% CI (3.34, 7.32), P < 0.0001]. The rates of proptosis [MD = 0.33, 95% CI (- 1.80, 2.46), P = 0.76] and diplopia [OR = 1.20, 95% CI (0.38, 3.76), P = 0.76] did not differ between patients who underwent medial inferior wall decompression and those who underwent balanced decompression. CONCLUSION: Balanced decompression and medial inferior wall decompression are both effective options for surgical treatment of thyroid-associated ophthalmopathy in clinical practice.


Subject(s)
Exophthalmos , Graves Ophthalmopathy , Humans , Graves Ophthalmopathy/surgery , Diplopia , Ophthalmologic Surgical Procedures , Decompression , Randomized Controlled Trials as Topic
20.
Am J Ophthalmol ; 262: 134-140, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38237748

ABSTRACT

PURPOSE: To clarify the clinical, diagnostic, and treatment characteristics of orbital liposarcoma. DESIGN: Retrospective observational case series. METHODS: A review was performed of electronic medical records, histopathology, radiological images, and follow-up information for 21 patients with orbital liposarcoma. RESULTS: The predominant clinical manifestation of this disease was painless exophthalmos. The most frequently encountered pathological types were well-differentiated and myxoid liposarcoma. Preoperative radiological images from 15 patients showed that orbital liposarcoma initially developed in extraocular muscle in 9 patients. Furthermore, all well-differentiated liposarcomas exhibited the radiographic characteristics of irregular and ill-defined adipose tissue, whereas only 12.5% of myxoid liposarcomas had the imaging characteristics. For the patients who exclusively underwent surgery, all of those with subtotal excisions experienced recurrence, 63.6% of marginal excisions recurred, and 50% of wide excisions resulted in recurrence. However, none of the patients who underwent marginal excisions or wide excisions combined with adjuvant radiotherapy exhibited recurrence. The analysis of magnetic resonance imaging findings in 3 patients who underwent neoadjuvant radiotherapy revealed that the tumor size remained stable in a patient with well-differentiated liposarcoma, whereas another patient with the same type of tumor exhibited a gradual increase in size. Conversely, a patient with myxoid liposarcoma experienced a significant reduction in tumor size following neoadjuvant radiotherapy. CONCLUSION: Orbital liposarcoma demonstrated a propensity for developing in the extraocular muscle. The radiological images of orbital well-differentiated liposarcomas were characterized by irregular and ill-defined adipose tissue. Surgery combined with radiotherapy demonstrated potential in reducing recurrence rates. Notably, orbital myxoid liposarcoma exhibited greater sensitivity to radiotherapy compared to well-differentiated liposarcoma.


Subject(s)
Liposarcoma , Magnetic Resonance Imaging , Orbital Neoplasms , Humans , Retrospective Studies , Orbital Neoplasms/diagnosis , Orbital Neoplasms/surgery , Orbital Neoplasms/therapy , Male , Female , Middle Aged , Aged , Adult , Liposarcoma/diagnosis , Liposarcoma/surgery , Liposarcoma/therapy , Tomography, X-Ray Computed , Ophthalmologic Surgical Procedures , Neoplasm Recurrence, Local , Aged, 80 and over , Follow-Up Studies , Exophthalmos/diagnosis , Oculomotor Muscles/surgery , Oculomotor Muscles/pathology , Oculomotor Muscles/diagnostic imaging , Radiotherapy, Adjuvant
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