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International Journal of Molecular... Jun 2024The gut microbial and metabolic characteristics of intestinal Behçet's disease (BD), a condition sharing many clinical similarities with ulcerative colitis (UC) and...
The gut microbial and metabolic characteristics of intestinal Behçet's disease (BD), a condition sharing many clinical similarities with ulcerative colitis (UC) and Crohn's disease (CD), are largely unexplored. This study investigated the gut microbial and metabolic characteristics of intestinal BD as well as potential biomarkers, comparing them with those in UC, CD, and healthy controls. Colon tissue and stool samples from 100 patients (35 UC, 30 CD, and 35 intestinal BD) and 41 healthy volunteers were analyzed using 16S ribosomal RNA sequencing to assess microbial diversity, taxonomic composition, and functional profiling. Plasma metabolomic analyses were performed using gas chromatography and ultra-performance liquid chromatography-mass spectrometry. Results indicated reduced microbial diversity in CD but not in intestinal BD, with intestinal BD showing fewer changes compared to controls yet distinct taxonomic features from UC, CD, and controls. Common alterations across all diseases included a reduction in beneficial bacteria producing short-chain fatty acids. Intestinal BD-specific changes featured a decreased abundance of Bacteroides fragilis. Metabolomic profiles in intestinal BD were similar to those in CD but distinct from those in UC, displaying significant changes in energy metabolism and genetic information processing. This integrative analysis revealed both shared and unique profiles in intestinal BD compared with UC, CD, and controls, advancing our understanding of the distinctive features of these diseases.
Topics: Humans; Behcet Syndrome; Metabolome; Gastrointestinal Microbiome; Male; Female; Adult; Middle Aged; RNA, Ribosomal, 16S; Crohn Disease; Metabolomics; Inflammatory Bowel Diseases; Biomarkers; Feces; Colitis, Ulcerative; Case-Control Studies
PubMed: 38928402
DOI: 10.3390/ijms25126697 -
BMC Neurology Jun 2024We present a rare case of NeuroBehcet's-related intracranial hypertension without cerebral venous thrombosis (NBrIHwCVT), occurring as the first presentation of... (Review)
Review
BACKGROUND
We present a rare case of NeuroBehcet's-related intracranial hypertension without cerebral venous thrombosis (NBrIHwCVT), occurring as the first presentation of NeuroBehcet's. In addition, we describe the novel use of subcutaneous tocilizumab for this indication. This is followed by a review of the literature on this topic.
CASE
The patient was a 28-year-old lady of Southern Chinese origin with a known history of Behcet's disease with oral ulcers and ocular findings for which she was on mycophenolate mofetil and adalimumab. She presented with a headache and bilateral disc swelling associated with an intracranial pressure (ICP) of > 40cmH20. There were no structural lesions or cerebral venous thrombosis (CVT) on imaging. Initial lumbar puncture had raised leucocytes and protein. We discuss diagnostic challenges given persistently elevated ICP despite subsequent non-inflammatory cerebrospinal fluid (CSF) profiles and non-response to acetazolamide. She eventually showed a response to immunosuppressant therapy in the form of pulsed methylprednisolone, cyclophosphamide and subsequently subcutaneous tocilizumab, supporting the diagnosis of NBrIHwCVT. Complete normalization of ICP remains challenging. Her disease course was severe, unusual for her ethnicity.
LITERATURE REVIEW
We identified 34 patients (including ours) from 14 publications. We found that the majority of NBrIHwCVT patients were young (average age of 34 years), with a slight female preponderance. Of the 17 cases in the literature with available data on CSF profile, none had raised leucocytes whilst one patient had elevated protein. Patients were generally treated with steroids and occasionally azathioprine, in line with the suspected autoimmune pathophysiology. Of 22 patients with data on outcome, six (27%) were noted to have recurrence of symptoms generally occurring a few months later.
CONCLUSION
As demonstrated by this case, NBrIHwCVT can present with BD with raised ICP even if there is no prior history of NB, central Asian ethnicity, cerebral venous thrombosis or features of inflammation on the CSF. We demonstrated how novel use of Tocilizumab may have a role in the management of NBrIHwCVT. Based on our literature review, patients were more likely to be young, female, display a non-inflammatory CSF picture, be treated with steroids and harbour a possibility of recurrence.
Topics: Humans; Female; Adult; Intracranial Hypertension; Behcet Syndrome; Antibodies, Monoclonal, Humanized; Immunosuppressive Agents
PubMed: 38877431
DOI: 10.1186/s12883-024-03708-x -
Journal of Ophthalmic Inflammation and... Jun 2024To report a case of Purpureocillium lilacinum endophthalmitis.
PURPOSE
To report a case of Purpureocillium lilacinum endophthalmitis.
METHODS
The case of a fungal endophthalmitis caused by Purpureocillium lilacinum documented in an immunocompetent patient with no apparent trigger.
RESULTS
A 64-year-old male with a two-month history of panuveitis in his left eye was referred to our hospital. Initially misdiagnosed as sympathetic ophthalmia due to a previous surgery on his right eye 4 months before the onset of the left ocular picture, the patient received corticosteroid treatment, leading to a rapid deterioration of the left eye condition. An urgent exploratory vitrectomy was performed to identify the underlying cause, revealing endophthalmitis. Microbiological investigation yielded Purpureocillium lilacinum as the causative agent. Despite intensive treatment, including intravitreal antibiotics and antifungals, along with another surgical intervention, clinical evolution remained unfavourable, ultimately leading to the evisceration of the affected eye.
CONCLUSIONS
Purpureocillium lilacinum poses a rare yet sever threat as a causative agent of fungal endophthalmitis. Managing such cases is challenging due to the delayed identification, fungus's resistance to common antifungals, and its association with prior corticosteroid misuse in most patients. This case underscores the crucial importance of heightened clinical suspicion, early diagnosis, and the exploration of alternative treatment strategies in addressing Purpureocillium lilacinum endophthalmitis. The challenges posed by this rare fungal pathogen emphasize the need for a multidisciplinary approach and continued research to improve outcomes in these complex cases.
PubMed: 38867117
DOI: 10.1186/s12348-024-00412-2 -
The Journal of Dermatological Treatment Dec 2024The incidence of cutaneous paradoxical reactions associated with IL-17 inhibitors has gained attention in recent literature. Our report aims to investigate the... (Review)
Review
The incidence of cutaneous paradoxical reactions associated with IL-17 inhibitors has gained attention in recent literature. Our report aims to investigate the characteristics of one rare paradoxical reaction, presenting as Behcet's disease. We reported one case of Behcet's-like disease induced by secukinumab in a patient with psoriasis. This patient, a young woman with a long history of psoriasis, showed significant improvement in her psoriatic condition after receiving four doses of secukinumab. Unexpectedly, she developed symptoms such as high fever, painful oral and genital ulcers, facial maculopapules, and erythema nodosum-like lesions on her lower limbs. Despite neutrophilia, there was no evidence of infection found in her laboratory tests. Histological analysis of a skin biopsy highlighted subcutaneous panniculitis and a mixed inflammatory cell infiltrate in the dermis. The patient was consequently diagnosed with secukinumab-induced Behcet's-like disease. Additionally, we have reviewed nine other documented cases of Behcet's-like disease triggered by IL-17 inhibitors. This group showed no significant gender preference, suffering from conditions such as psoriasis, ankylosing spondylitis, and hidradenitis suppurativa. Oral and genital ulcers were prevalent among the paradoxical reactions noted. Marked improvement was observed in all patients upon discontinuation of the IL-17 inhibitors. Our report serves to alert physicians to this uncommon but significant paradoxical effect that may arise with anti-IL-17 treatment.
Topics: Humans; Female; Antibodies, Monoclonal, Humanized; Behcet Syndrome; Psoriasis; Adult; Interleukin-17; Skin
PubMed: 38857894
DOI: 10.1080/09546634.2024.2347440 -
Journal of Medical Case Reports Jun 2024To report a case of Multiple Evanescent White Dot Syndrome (MEWDS) one month after a COVID-19 infection in a female patient at an age unusual for the occurrence of this...
BACKGROUND
To report a case of Multiple Evanescent White Dot Syndrome (MEWDS) one month after a COVID-19 infection in a female patient at an age unusual for the occurrence of this disease.
CASE PRESENTATION
A 69-year-old Caucasian female reported the presence of floaters, photopsia, and enlarging vision loss in her left eye following the COVID-19 infection. Clinical and multimodal imaging was consistent with the MEWDS diagnosis. Fluorescein angiography examination revealed characteristic hyperfluorescent spots around the fovea in a wreath-like pattern. An extensive lab workup to rule out other autoimmune and infectious etiologies was inconclusive. Visual acuity and white dots resolved after a course of corticosteroids, which was confirmed on follow-up dilated fundus exam and multimodal imaging.
CONCLUSIONS
MEWDS is a rare white dot syndrome that may occur following COVID-19 infection in addition to other reported ophthalmic disorders following this infection.
Topics: Humans; Female; COVID-19; Aged; Fluorescein Angiography; SARS-CoV-2; Tomography, Optical Coherence; White Dot Syndromes; Visual Acuity; Retinal Diseases; Vision Disorders
PubMed: 38849848
DOI: 10.1186/s13256-024-04596-y -
BMC Ophthalmology Jun 2024Several immune checkpoint inhibitors (ICIs) have been linked to the occurrence of Vogt-Koyanagi-Harada disease (VKHD)-like uveitis. Among the ICIs, there has been no...
BACKGROUND
Several immune checkpoint inhibitors (ICIs) have been linked to the occurrence of Vogt-Koyanagi-Harada disease (VKHD)-like uveitis. Among the ICIs, there has been no report of immune-related adverse events (irAEs) caused by a new programmed death protein-1(PD-1) monoclonal antibody (Toripalimab).
CASE PRESENTATION
This paper presents a case of VKHD-like uveitis that arose following Toripalimab therapy for urothelial cancer of the bladder, and the patient experienced symptoms 10 days after the final dosage of 20 months of medication treatment. This patient with bladder uroepithelial carcinoma had severe binocular acute panuveitis with exudative retinal detachment after receiving Toripalimab therapy. Binocular VKHD-like uveitis was suggested as a diagnosis. Both eyes recovered after discontinuing immune checkpoint inhibitors and local and systemic corticosteroid treatment.
CONCLUSIONS
This report suggests that VKHD-like uveitis can also occur in patients receiving novel PD-1 antibodies and the importance of paying attention to eye complications in patients receiving treatment over a long period.
Topics: Humans; Uveomeningoencephalitic Syndrome; Immune Checkpoint Inhibitors; Male; Uveitis; Urinary Bladder Neoplasms; Programmed Cell Death 1 Receptor; Antibodies, Monoclonal, Humanized; Female; Middle Aged; Aged; Antineoplastic Agents, Immunological
PubMed: 38849786
DOI: 10.1186/s12886-024-03484-9 -
Cureus May 2024We report four cases of syphilitic uveitis with diverse clinical presentations. All patients were men who have sex with women, and were aged 19-68 years, and none were...
We report four cases of syphilitic uveitis with diverse clinical presentations. All patients were men who have sex with women, and were aged 19-68 years, and none were HIV-positive. All cases were bilateral. One case presented with anterior uveitis, while three exhibited panuveitis. One patient had acute syphilitic posterior placoid chorioretinitis and two had retinal vasculitis resulting in damage to the outer retinal and retinal pigment epithelium. The rapid plasma reagin (RPR) test and (TP) hemagglutination test were both positive in all cases. Six of eight eyes had improved vision and best-corrected visual acuity better than 20/20 after antibiotic treatment. Serological testing is mandatory for the diagnosis of syphilitic uveitis. Additionally, multimodal imaging, including optical coherence tomography (OCT), fundus autofluorescence (FAF), and fluorescein angiography (FA), can provide useful adjunctive information for early diagnosis and assessment of treatment response.
PubMed: 38846191
DOI: 10.7759/cureus.59791 -
BMC Ophthalmology May 2024Several autoimmune disorders have been linked to polymorphisms in IL10 and IL6R genes. This research aimed to study whether single nucleotide polymorphisms (SNPs) in the...
BACKGROUND
Several autoimmune disorders have been linked to polymorphisms in IL10 and IL6R genes. This research aimed to study whether single nucleotide polymorphisms (SNPs) in the genes of IL10 and IL6R were associated with acute anterior uveitis (AAU) in Han Chinese.
METHODS
Genotyping was carried out by the iPLEX Gold Genotyping Assay. Our study comprised 420 patients with AAU and 918 healthy subjects from Han Chinese. Using the chi-square (χ2) test, alleles and genotypes were analyzed between AAU subjects and healthy controls.
RESULTS
All ten SNPs were successfully genotyped and four SNPs (IL10/rs1800871, IL10/rs3021094, IL10/rs2222202, IL6R/rs4845618) exhibited weak associations with AAU, as indicated by their P values. However, upon applying the Bonferroni correction, there was no significant association between AAU and the control subjects. Additionally, the haplotype analysis of the ten SNPs revealed no association with AAU.
CONCLUSION
Our findings suggested that polymorphisms of the tested ten SNPs on the IL10 and IL6R genes did not show any association with the risk of developing AAU among the Han Chinese population.
Topics: Humans; Uveitis, Anterior; Male; Interleukin-10; Female; Receptors, Interleukin-6; Polymorphism, Single Nucleotide; Adult; China; Acute Disease; Middle Aged; Asian People; Genotype; Genetic Predisposition to Disease; Case-Control Studies; Gene Frequency; Young Adult; Alleles; Haplotypes; Aged; East Asian People
PubMed: 38822340
DOI: 10.1186/s12886-024-03495-6 -
Scientific Reports May 2024The advent of coronavirus disease 2019 (COVID-19) pandemic has affected the incidence and course of various diseases and numerous studies have investigated ocular...
The advent of coronavirus disease 2019 (COVID-19) pandemic has affected the incidence and course of various diseases and numerous studies have investigated ocular involvement associated with COVID-19 and corresponding vaccines. In this study, we compared the incidence of multiple evanescent white dot syndrome (MEWDS) before and during the COVID-19 pandemic at a single center in Korea and analyzed the demographic and clinical features of patients with MEWDS presenting during the COVID-19 pandemic. We categorized patients with MEWDS into two groups according to date of diagnosis. Pre-COVID19 group included patients diagnosed during the pre-pandemic period (between March 11, 2017, and March 10, 2020), whereas post-COVID19 group included patients diagnosed during the pandemic period (between March 11, 2020, and March 10, 2023). 6 and 12 patients were included in pre-COVID19 group and post-COVID19 group, respectively. Among all hospital visits during the pre-pandemic and pandemic periods, 0.011% and 0.030% were due to MEWDS, indicating a significant increase during the pandemic (p = 0.029, B = 2.756). The annual incidence of patients with MEWDS in 2017-2022 were 0.73, 0.75, 0.78, 1.32, 2.49, and 2.07 per 10,000 population, respectively, corresponding to a significant increase (p = 0.039, B = 1.316). Our results imply that the incidence and manifestation of MEWDS are likely to become more diverse in the COVID-19 pandemic era.
Topics: Humans; COVID-19; Male; Female; Incidence; Republic of Korea; Middle Aged; Adult; SARS-CoV-2; White Dot Syndromes; Aged; Pandemics
PubMed: 38816508
DOI: 10.1038/s41598-024-63255-w -
Indian Journal of Ophthalmology Jun 2024Leptospirosis is a waterborne zoonotic disease prevalent in tropical regions, causing significant morbidity and mortality. It can involve any organ in its primary stage,...
PURPOSE
Leptospirosis is a waterborne zoonotic disease prevalent in tropical regions, causing significant morbidity and mortality. It can involve any organ in its primary stage, and uveitis is its late complication. While advanced laboratory diagnosis is available only in tertiary care centers globally, a cost-effective bedside assessment of clinical signs and their scoring could offer a provisional diagnosis.
AIM
To analyze the diagnostic potential of demographic and clinical signs in a large cohort of serologically confirmed leptospiral uveitis patients.
METHODS
In this retrospective study, demographic and clinical parameters of 876 seropositive leptospiral uveitis patients and 1042 nonleptospiral uveitis controls were studied. Multivariable logistic regression analysis with bootstrap confidence interval (CI) characterized the diagnostic predictors. The performance of the model was evaluated using the area under the receiver operating curve (AUROC).
RESULTS
Presence of nongranulomatous uveitis (odds ratio [OR] = 6.9), hypopyon (OR = 4.6), vitreous infiltration with membranous opacities (OR = 4.3), bilateral involvement (OR = 4), panuveitis (OR = 3.3), vasculitis (OR = 1.9), disc hyperemia (OR = 1.6), absence of retinochoroiditis (OR = 15), and absence of cystoid macular edema (OR = 8.9) emerged as predictive parameters. The AUROC value was 0.86 with 95% CI of 0.846-0.874. At a cut-off score of 40, the sensitivity and specificity were 79.5 and 78.4, respectively.
CONCLUSION
The study demonstrates that ocular signs can serve as diagnostic predictors for leptospiral uveitis, enabling primary care ophthalmologists to make bedside diagnosis. This can be further confirmed by laboratory methods available at tertiary care centers.
Topics: Humans; Retrospective Studies; Leptospirosis; Male; Female; Eye Infections, Bacterial; Uveitis; Adult; Leptospira; Middle Aged; ROC Curve; Young Adult; Adolescent
PubMed: 38804803
DOI: 10.4103/IJO.IJO_1376_23