Bilateral Inflammation Research Articles

Reversible cataract after exposure to distilled water: a case report

BackgroundTo report a case of a reversible cataract after intracameral infusion of distilled water during full-thickness astigmatic keratotomy.Case presentationA 20-year-old male whose bilateral anterior chambers were exposed to distilled water during astigmatic keratotomy developed bilateral corneal edema, inflammation of the anterior chambers, and an anterior subcapsular cataract in the right eye. After 1 month of topical administration of 0.1% prednisolone acetate and 5% NaCl, the bilateral inflammation of the cornea and anterior chambers cleared; endothelial cell density decreased by 41.1% in the right eye and 12.7% in the left eye. The cataract in the right eye decreased centripetally. Small incision lenticule extraction surgery was performed at 2 months after the astigmatic keratotomy; the patient’s uncorrected distance visual acuity 3 months later was 20/25 in both eyes.ConclusionsThis case suggests that a cataract that develops as a result of instantaneous intracameral exposure to distilled water is reversible.

ZASTOSOWANIE ZACZEPU NA ODNOGĘ KRÓTKĄ KOWADEŁKA IMPLANTU UCHA ŚRODKOWEGO VIBRANT SOUNDBRIDGE W PRZEWLEKŁYM ZAROSTOWYM ZAPALENIU PRZEWODU SŁUCHOWEGO ZEWNĘTRZNEGO – OPIS PRZYPADKU

ObjectiveChronic obstructive inflammation of the external ear canal after different kinds of surgeries often leads to restenosis. There are different approaches to treatment of such cases, and one solution is a middle ear implant. Here we analyze the efficacy of using the Vibrant Soundbridge middle ear implant with a new incus short process (SP) coupler in this type of case.Material and MethodsWe present a case study of a 29-year old patient with bilateral mixed hearing loss caused by bilateral obstructive inflammation of the external ear canals. He had undergone meatoplasty with very short-lived improvement. Afterwards he qualified for middle ear implantation to restore hearing, and the Vibrant Soundbridge was implanted with a new SP coupler to which the floating mass transducer (FMT) is attached. Postoperative hearing tests included pure tone audiometry, free field audiometry, speech recognition, and APHAB questionnaire.ResultsSpeech recognition after implantation improved to 100%. Pre- and postoperative bone conduction thresholds were unchanged. Vibrometry thresholds 10 months after surgery did not deteriorate. There was a decrease in global APHAB score by 61% from 2 to 10 months after implantation.ConclusionsApplication of the Vibrant Soundbridge middle ear implant can be an effective treatment in cases of hearing loss caused by obstructive inflammation of the external ear canals. The new incus SP coupler seems to be an effective method of stimulating the middle ear in mixed hearing loss.

Enhanced interferon gamma response contributes to disease remission in Rheumatoid Arthritis.

Abstract Rheumatoid arthritis (RA) is an autoimmune disease with debilitating bilateral inflammation in multiple joints. Although newer biologics are very effective in managing RA, critical gaps in immunopathogenic mechanism remain. We recently showed that increased expression of the interferon gamma (IFN-γ) receptor in PBMC associated with RA and increased radiographic severity. We hypothesized that enhanced IFN-γ signaling in specific subpopulations of mononuclear cells from patients with RA contributes to disease severity. To test this, we collected PBMC from healthy controls (HC) and RA patients in remission, low disease and moderate disease severity as per American College Criteria. As an autoimmune disease control, we compared PBMC from treatment naïve multiple sclerosis (MS) patients. We quantitated classical (JAK/STAT) and non-classical (ERK/MTOR) IFN-γ induced signaling in subpopulations of T and B cells within PBMC using phospho-flow cytometry. For analysis we utilized t-SNE and SPADE data visualization and clustering tools to interrogate changes in activation and marker expression within and between T and B cell populations. We observed IFN-γ-induced activation of STAT1 in CD4+ naïve and central memory T-cells from was greatest in RA patients in remission and lowest in individuals with moderate disease. IFN-γ induced activation of STAT1 in all CD8+ T-cell sub-populations of RA were similar to that from HC. Furthermore, the activation of STAT1 in all CD4+ T-cell sub-populations in PBMC from MS patients very generally greater than from RA patients and HC. From our results we infer that that while expression of IFNGR correlates with disease in RA, gain in response to IFN-γ is indicative of treatment response and RA remission.

Bilateral Idiopathic Orbital Inflammation Syndrome in an adult patient: A rare case report

Bilateral Idiopathic Orbital Inflammation Syndrome (IOIS) is rare in adults. Most commonly it affects the young adults and children. It is mostly unilateral condition. This is a case of 50 years old male coming with the complaints of painless proptosis on both the eyes for 1 year. The history of diplopia was also found. On examination, it was found that there was restriction of movements in Right eye superiorly. Any history of associated systemic illness was not found. Based on the clinical examination, laboratory reports and contrast enhanced computed tomography (CECT) the diagnosis of Bilateral IOIS was made. Patient was treated with Intravenous methyl prednisolone followed by oral Prednisolone in tapering doses for a period of 2 months. After 2 month of regular follow up the patient was found to have a favorable outcome with improved visual acuity and reduction in the size of the proptosed eye. Patient was followed up for another 6 month to observe any recurrence or relapse.

Adrenocorticotropic hormone analogue as novel treatment regimen in ocular cicatricial pemphigoid

PurposeTo report the clinical outcome of a patient with ocular cicatricial pemphigoid, treated with adrenocorticotropic hormone gel. ObservationsA 75-year-old female with a biopsy proven ocular cicatricial pemphigoid (OCP) presented with bilateral conjunctival inflammation, fornix shortening, subepithelial fibrosis and corneal scarring. The patient was previously treated with topical steroids, topical cyclosporine and lubricating drops, and had undergone several amniotic membrane transplants due to recurrent corneal erosions. Once OCP diagnosis was established, the patient was started on oral corticosteroids (60 mg daily). In order to wean the patient off from systemic steroids, other immunomodulatory agents had been tried, including mycophenolate mofetil (1000 mg twice daily) and methotrexate (up to 25 mg weekly). However, none of these agents adequately controlled the ocular surface inflammation, and the patient experienced bilateral progressive cicatrization and corneal decompensation, as well as the development of side effects from the systemic corticosteroids, methotrexate and mycophenolate mofetil therapies. Treatment with twice weekly subcutaneous adrenocorticotropic hormone (ACTH) gel was initiated, along with tapering of systemic corticosteroids. During the 19 months treatment period, the patient demonstrated significant improvement in the ocular surface inflammation, visual acuity was stable and no significant adverse effects were observed. Systemic corticosteroids dosage was successfully reduced from 10 mg/day to none at last follow up. Conclusions and importanceACTH gel has shown to be an effective and safe treatment option for chronic, refractory and progressive ocular inflammatory disease. To the best of our knowledge, this is the first case report of a patient with OCP, treated successfully with ACTH gel. This case report may encourage ophthalmologists to employ ACTH gel in the management of OCP.

Birdshot Chorioretinopathy treated with dexamethasone intravitreal implant and a biologic response modifier: a successful management

Birdshot chorioretinopathy is an organ-specific, potentially blinding chronic of bilateral inflammation of the posterior eye segment. It is seen mainly in persons who have the HLA-A29 haplotype. The diagnosis remains clinical and is based in his hallmark, which is the presence of multiples white dots throughout the fundus, resembling a shotgun pattern. The immune mechanisms involved in its pathogenesis remains unclear and the long-term outcome is guarded. However, the use of immunosuppressive agents and newer therapies demonstrated control of the inflammation and is a hope mainly in refractory cases. We report a case of Birdshot chorioretinopathy treated successfully with steroids and immunosuppressive agents.

Panuveitis simulating ocular Behçet’s in cases of chronic myelogenous leukaemia in remission

We report two patients with chronic myelogenous leukaemia (CML) in remission phase who developed panuveitis simulating Behçet’s disease. A 26-year-old man presented with bilateral panuveitis (hypopyon in the right eye,...

Silent tracheobronchial chondritis in a patient with a delayed diagnosis of relapsing polychondritis

Relapsing polychondritis is a very rare autoimmune disease characterised by a relapsing inflammation of hyaline, elastic and fibrous cartilaginous tissues. The incidence is estimated to be between 3.5 and 4.5...

Relapsing Polychondritis Initially Presenting with Hoarseness and Difficulty Breathing in a 21-Year-Old Male

Objective: To present a case of relapsing polychondritis initially presenting with hoarseness and difficulty breathing and to discuss the diagnostic criteria and typical CT scan findings of relapsing polychondritis.
 Design: Case Report
 Setting: Tertiary Private Hospital in Metro Manila
 Case Report: A 21-year-old man who was initially managed as a case of bronchial asthma for persistent hoarseness and recurrent difficulty breathing was found to have severe laryngeal edema on endoscopy, and soft tissue expansion of the cricoid cartilage with calcifications and irregular first tracheal ring on CT scan. He also had recurrent eye redness and developed bilateral aural inflammation, and was subsequently diagnosed to have relapsing polychondritis.
 Conclusion: Relapsing polychondritis is a rare autoimmune disease characterized by recurrent inflammation and eventual destruction of cartilage throughout the body. Typical manifestations may not always be present, causing a delay in diagnosis. It should be considered in patients with intractable respiratory symptoms not responsive to treatment for upper respiratory tract infections or asthma. A CT scan may reveal signs of cartilage destruction and help in diagnosis. 
 Keywords: Polychondritis, Relapsing; airway involvement; imaging

Inhibition of CD8+ T cells and elimination of myeloid cells by CD4+ Foxp3- T regulatory type 1 cells in acute respiratory distress syndrome.

Acute lung injury and acute respiratory distress syndrome (ARDS) are caused by rapid-onset bilateral pulmonary inflammation. We therefore investigated the potential role of interleukin (IL)-10+ CD4+ Tr1 cells, a regulatory T cell subset with previously identified immunosuppressive functions, in ARDS patients. We first showed that circulating Tr1 cells were upregulated in active and resolved ARDS patients compared to healthy controls and pneumonia patient controls. A significant fraction of these Tr1 cells expressed granzyme B and perforin, while most Tr1 cells did not express interferon gamma (IFN-γ), IL-4, IL-17 or FOXP3, suggesting that the effector functions of these Tr1 cells were primarily mediated by IL-10, granzyme B, and perforin. Indeed, Tr1 cells effectively suppressed CD8+ T cell IFN-γ production and induced lysis of monocytes and dendritic cells in vitro. The elimination of myeloid antigen-presenting cells depended on granzyme B production. We also discovered that Tr1 cells could be identified in the bronchoalveolar lavage fluid collected from ARDS patients. All these results suggested that Tr1 cells possessed the capacity to downregulate inflammation in ARDS. In support of this, we found that ARDS patients who resolved the inflammation and survived the syndrome contained significantly higher levels of Tr1 cells than ARDS patients who succumbed to the syndrome. Overall, this report added a novel piece of evidence that ARDS could be intervened by regulatory T cell-mediated suppressive mechanisms.

Corneal Endothelium in Patients with Anterior Uveitis

To investigate a possible effect of intraocular inflammation on corneal endothelium by describing corneal endothelial cell density (ECD) and morphologic variables in eyes with anterior uveitis, and to investigate factors that may influence these findings. Cross-sectional, observational study. Observers were not masked. Volunteers with histories of unilateral or bilateral anterior segment inflammation (anterior, intermediate, or panuveitis); included were 52 patients (84 eyes with uveitis). Endothelial cell density and morphologic variables of both eyes of all study participants were determined by specular microscopy; central corneal thickness was determined by ultrasound pachymetry. Central corneal ECD, coefficient of variability, percentage hexagonality, and central corneal thickness. Central ECD was lower among eyes that had undergone cataract or glaucoma surgery or both (n=28; P= 0.0004). After exclusion of eyes with surgery, variables for eyes with uveitis (n= 56) were compared with 2 historical populations of normal, age-matched controls and with contralateral eyes in individuals with unilateral uveitis. Central ECD was lower in eyes with uveitis than in control eyes for all age groups (P ≤ 0.01 for four of six 10-year age intervals compared with the primary control group). Among patients with unilateral uveitis who had not undergone surgery in either eye (n= 12), central ECD was lower in eyes with uveitis (2324 cells/mm(2) [range, 1543-3289 cells/mm(2)]) than in contralateral eyes (2812.5 cells/mm(2) [range, 1887-3546 cells/mm(2)]; P=0.0005), and percentage hexagonality was lower in eyes with uveitis (54% [range, 33%-66%]) than in contralateral eyes (58.5% [range, 52%-82%]; P= 0.004). There was no significant difference in central corneal thickness between eyes with and without uveitis (P= 0.27). No eyes had clinically apparent central corneal edema. Relationships remained unchanged after exclusion of eyes with herpetic anterior uveitis. Host and disease-related characteristics were evaluated as risk factors for variations in outcome measures. Central ECD was correlated to the duration of active uveitis (r=-0.41; P < 0.0001), maximum intraocular pressure during the course of disease (r=-0.40; P= 0.0002), and maximum laser flare photometry value (r=-0.26; P= 0.020). Observed relationships suggest that anterior segment inflammation adversely affects the corneal endothelium. Longitudinal studies are warranted to determine whether long-standing anterior uveitis increases risk of endothelial dysfunction, especially in the setting of intraocular surgery.

Bilateral Orbital Inflammation with Optic Neuropathy as a Paraneoplastic Syndrome Associated with Non-small Cell Lung Cancer

Bilateral Orbital Inflammation with Optic Neuropathy as a Paraneoplastic Syndrome Associated with Non-small Cell Lung Cancer

Abstract 14184: Patients With Coronary Artery Disease and Bilateral Carotid Inflammation Show Higher One Year Rate of Cardiovascular Events

Background: Inflammation is an essential characteristic of vulnerable atherosclerotic plaque. Although its prognostic value during invasive assessment has been documented, the clinical significance of its routine evaluation is still under dispute. Microwave radiometry (MWR) allows the rapid, in vivo, noninvasive assessment of the internal temperature of carotid arteries, reflecting local inflammation. The aim of the present study was to evaluate the prognostic role of carotid atherosclerotic plaque temperatures in patients with documented coronary artery disease (CAD). Methods: Consecutive patients with significant CAD as documented by coronary angiography were included in the study. All patients underwent evaluation of both carotid arteries by 1) ultrasound and 2) microwave radiometry (MWR). During ultrasound study maximum carotid plaque thickness was assessed in all carotids. Temperature difference (ΔT) by MWR was defined as maximal temperature along the carotid artery minus minimum. ΔT ≥0.90°C was assigned as high ΔT. Major cardiovascular event (MACE) was defined as death, stroke, myocardial infarction or revascularization. All patients were followed-up clinically for one year. Results: In total 192 patients were included in the study. At baseline thirty patients (15.6%) had high ΔT temperatures (≥0.90°C) in both carotid arteries. Max plaque thickness was slightly higher in this group (2.75±1.34 vs 2.27±1.03, p=0.07). MACE was 23.3% in the group with bilateral high ΔT and 4.3% in non-high ΔT group (p&lt;0.001). By multivariate logistic regression analysis, ΔT was an independent predictor for MACE, when adjusted for sex, age contemporary risk factors, number of vessels with significant stenosis and maximum carotid plaque thickness (OR: 6.10, 95% CI 1.56-23.80, p=0.001). In Kaplan-Meier plots patients with bilateral high ΔT showed higher event and mortality rates, compared with patients in non-high ΔT group (log-rank p=0.001 for both comparisons). Conclusion: The presence of bilateral high carotid artery temperatures as assessed by MWR has implications in short term prognosis of patients with CAD. A long term study is warranted to establish the prognostic impact of MWR in this high-risk population.

Choroidal neovascularization secondary to tubulointerstitial nephritis and uveitis syndrome (TINU) in an adult patient.

BackgroundInflammation is a well-known risk factor for the development of choroidal neovascularization (CNV), yet not all causes of intraocular inflammation have been documented to cause CNV. Tubulointerstitial nephritis and uveitis syndrome (TINU) is a rare cause of intraocular inflammation mostly in pediatric patients and only seldom has been associated with development of CNV.FindingsA 34-year-old pregnant female with a past history of bilateral ocular inflammation secondary to TINU presents 1 year after diagnosis with vision loss in the left eye. Clinical examination and investigations show the development of CNV in the left eye. The patient was treated with ranibizumab (Genentech, San Francisco, CA) intravitreal injections with improvement in symptoms and clinical findings.ConclusionsWe report the first case of CNV secondary to TINU in an adult patient. The CNV associated with TINU is responsive to intravitreal anti-vascular endothelial growth factor (anti-VEGF) therapy.

Sympathetic ophthalmia within the limit of the radical surgical recommendation - forensic implications

Sympathetic ophthalmia is a bilateral diffuse granulomatous inflammation of the surgical or non-surgical eye trauma that occur as a rare and severe complication of the trauma (1:14,000). The injured eye becomes the excitatory eye that initiates the inflamatory state of the non-injured eye that becomes the sympathetic eye.<br/> The conservative therapeutic approach, i.e. the medical treatment (steroid and non steroidal anti-inflammatory therapy and immunomodulatory therapy) is recognized as the first choice compared to the radical treatment which is injured eye evixceration. However, the professional experience and the clinic evolution may play an important role. Clinical practice brings cases at the border of radical and conservative treatment with complex ethical and legal implications that are discussed. We present two cases which received a medical approach in one case and a surgical approach in other case. Based on our experience we consider that the only true prophylaxis for the loss of a healthy eye is the enucleation of the traumatized eye at the very first symptoms: many ophtalmologists share our vision. Evisceration also, in selected cases, is an acceptable alternative. We discuss also about malpraxis and liability issues of such a professional reccomendation as much as ethical issues such as beneficence and non-maleficence which seems to be contradictory if we considered one eye vision or both eye vision as the best outcome.

Lubricity feature enhances SLK management

Purpose: Superior Limbic Keratoconjunctivitis (SLK) typically presents as an asymptomatic bilateral inflammation of the superior bulbar conjunctiva, associated with thyroid disease in woman aged 20–60. Although the pathophysiology of SLK is unknown, it is believed that swelling of the conjunctiva results in mechanical irritation/friction with the lid.

Sympathetic ophthalmia in a case of penetrating ocular injury associated with brain injury: A case report

Sympathetic ophthalmia is a rare bilateral granulomatous inflammation that follows accidental or surgical insult to the uvea of one eye. The diagnosis may be confounded by the associated traumatic brain injury. Here, we report a case of sympathetic opthalmia in a patient who sustained perforating injury of the right eye and traumatic brain injury in the left frontal area following a road traffic accident. Timely diagnosis and surgical intervention was the key to a successful outcome.

Bilateral steroid induced glaucoma in vernal keratoconjunctivitis

VKC is a bilateral recurrent allergic interstitial conjunctival inflammation with a periodic seasonal incidence and of self limiting nature, mainly affecting the younger population. Patients of VKC on steroid therapy are at higher risk of developing steroid induced glaucoma. Raised intraocular pressure due to steroids typically occurs within few weeks of starting steroid therapy and comes back to normal on immediate stoppage of steroids. A case of steroid induced glaucoma in a 30 years old female with vernal keratoconjunctivitis. She was on topical steroids for 3-4 years. She was incompliant with the instructions to stop steroids. She eventually developed steroid induced glaucoma and glaucomatous optic neuropathy with tunnel vision.

The immunopathogenesis of birdshot chorioretinopathy; a bird of many feathers.

Birdshot chorioretinopathy (BSCR) is a bilateral chronic intraocular inflammation or posterior uveitis that preferentially affects middle-aged Caucasians. BSCR is characterized by distinctive multiple choroidal hypopigmented lesions in combination with retinal vasculitis and vitritis, and the extraordinary feature that virtually all patients are HLA-A29 positive. Its pathophysiology is still poorly understood. BSCR is the strongest documented association between HLA and disease in humans, which makes it an excellent model for studying the underlying immuno-genetic mechanisms of HLA class I-associated diseases. Although the association with HLA-A29 suggests that it is directly involved in the presentation of peptide antigens to T cells, the exact contribution of HLA-A29 to the pathophysiology of BSCR remains enigmatic. This article revisits the HLA-A29 peptidome using insights from recent studies and discusses why HLA-A29 can be considered a canonical antigen presenting molecule. The first genome-wide association study facilitated novel concepts into a disease mechanism beyond HLA-A29 that includes strong genetic predisposition for the ERAP2 gene that affects antigen processing for HLA class I. Furthermore, patients manifest with pro-inflammatory cytokine profiles and pathogenic T cell subsets that are associated with IL-17-linked inflammation. We are beginning to understand that the underlying biology of BSCR comprises various pathologic aspects branched into multiple molecular pathways. We propose to employ Systems Medicine to reveal their dynamic interplay for a holistic view of the immunopathology of this intriguing archetypal HLA class I-associated disease.

English

Vernal keratoconjunctivitis (VKC) is a recurrent seasonal disease of childhood, characterized by severe bilateral inflammation of the conjunctiva and by giant papillae of the superior tarsal conjunctiva, gelatinous hypertrophy of the limbus, and keratopathy. Clinical and immunohistochemical studies suggest that IgE-dependent (type I allergic) and IgE-independent (type IV allergic) mechanisms are involved in the immunopathogenesis of VKC, in which various inflammatory cells, including different T cell subpopulations play an active role via a cascade of chemical mediators. Endocrine, genetic, neurogenic, environmental and socioeconomic risk factors have been identified. The clinical course of this disease is usually benign and self-limiting, but a minority of patients will face very debilitating and sight threatening complications. The variety of currently available drugs to treat VKC include anti-histamines, mast-cell stabilizers, dual acting agents, corticosteroids and immunomodulators but none is enough to treat all aspects of the multifaceted pathophysiology of VKC. More selective drugs like anti-chemokine receptor antibodies and leukotriene receptor antagonists are under evaluation. Cyclosporine has been shown to be effective in the treatment of VKC but further randomized control trials are required to establish the minimum effective concentration. The purpose of this paper is to describe the clinical expression of VKC, to discuss its pathogenic mechanisms, and to suggest novel therapeutic strategies.