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ocular cicatricial pemphigoid

Caiyun You, Neerav Lamba, Andres F Lasave, Lina Ma, Mikhail Hernandez Diaz, C Stephen Foster
PURPOSE: The purpose was to evaluate the effectiveness and safety of rituximab (RTX) for the treatment of patients with aggressive ocular cicatricial pemphigoid (OCP). METHODS: A review of patient records at a tertiary referral center with biopsy confirmed OCP who presented between 2006 and 2016. Sixty-one eyes of 32 patients with symptomatic OCP who received treatment with RTX monotherapy or RTX in combination with additional immunomodulatory treatment (IMT) were evaluated...
February 3, 2017: Graefe's Archive for Clinical and Experimental Ophthalmology
Kaveh Vahdani, Vladimir T Thaller, Giorgio Albanese, Andrew F Dean
PURPOSE: To describe an unusual cicatrizing manifestation of periocular amyloid and the utility of adjuvant antimetabolite therapy. METHODS: This is a case report of a 49-year old woman with progressive bilateral upper lid ptosis, who was found to have bilateral inferior forniceal masses with thickening of the conjunctiva. Conjunctival and eyelid biopsies showed evidence of amyloid deposition. Systemic evaluation did not reveal any evidence of systemic amyloidosis...
January 11, 2017: Cornea
Linda Yi-Chieh Poon, James Chodosh, Demetrios G Vavvas, Claes H Dohlman, Teresa C Chen
We describe the surgical technique of endoscopic cyclophotocoagulation in a Boston keratoprosthesis type II patient. This patient with ocular cicatricial pemphigoid had pars plana endoscopic cyclophotocoagula through wounds created in the eyelids.
April 2017: Journal of Glaucoma
Soheil Tavakolpour
BACKGROUND: Mucous membrane pemphigoid (MMP) is considered an autoimmune blistering disease that predominantly affects mucous membranes. Various treatments are available for controlling the diseases, but not all of them may respond. MATERIALS AND METHODS: PubMed and Google Scholar were searched for all the associated studies until 2015, using the keywords such as "cicatricial pemphigoid" or "ocular pemphigoid" or "mucous membrane pemphigoid" or "MMP" and "intravenous immunoglobulin" or "IVIg" to find all the relevant studies...
2016: Journal of Research in Medical Sciences: the Official Journal of Isfahan University of Medical Sciences
Bernhard Steger, Savitha Madhusudan, Stephen B Kaye, Amira Stylianides, Vito Romano, Sundas E Maqsood, Janice Harper, Sajjad Ahmad
PURPOSE: Despite the availability of systemic immunosuppressants, cicatricial conjunctivitis (CC) remains a potentially blinding ocular surface disease. We aim to describe the combined use of rituximab (RTX) and intravenous immunoglobulin (IVIg) for severe recalcitrant autoimmune CC. METHODS: In this single-center retrospective interventional case series with follow-up between 32 and 65 months, 3 cases with mucous membrane pemphigoid (patients 1-3) and 1 case with linear IgA disease (patient 4) were included...
December 2016: Cornea
C Günther
Autoimmune bullous diseases are characterized by intraepidermal or subepidermal autoantibody deposition that leads to blisters and secondary erosion. Mucous membranes are frequently affected in pemphigus vulgaris and always involved in cicatricial and mucosal pemphigoid. Mucosal lesions are detected less frequently in patients with bullous pemphigoid or epidermolysis bullosa acquisita. The diagnosis of autoimmune bullous disorders is based on determination of the subtype of autoantibodies bound in the skin and the clinical picture...
October 2016: Der Hautarzt; Zeitschrift Für Dermatologie, Venerologie, und Verwandte Gebiete
Qin Long, Ya-Gang Zuo, Xue Yang, Ting-Ting Gao, Jie Liu, Ying Li
AIM: To describe the clinical features and microstructural characteristics assessed by in vivo confocal microscopy (IVCM) in patients with ocular cicatricial pemphigoid (OCP). METHODS: A descriptive, uncontrolled case series study. Patients diagnosed with OCP were examined by clinical history, slit-lamp biomicroscopy features and IVCM images. The results of direct immunofluorescence (DIF) biopsies and indirect immunofluorescence (IIF) were also recorded. Local and systemic immunosuppressive therapy were administered and adjusted according to response...
2016: International Journal of Ophthalmology
E Szabó, M Palos, P Skalická
INTRODUCTION: Ocular cicatricial pemphigoid (OCP) is rare, severe, sight threatening autoimmune disease of the conjunctiva, which affects elderly patients, more often women. AIM: To evaluate the success rate of stabilisation of ocular findings in patients with OCP. METHODS: Retrospective study of patients from Centre of Conjunctival and Corneal Diseases at Department of Ophthalmology, General University Hospital and 1st Medical Faculty of Charles University in Prague in 1992-2013 was performed...
February 2016: Ceská a Slovenská Oftalmologie
Karen C Broussard, Theresa G Leung, Ahmadreza Moradi, Jennifer E Thorne, Jo-David Fine
Autoimmune blistering diseases are a heterogeneous group of disorders that mostly affect the skin and mucous membranes. Occasionally, other organ systems may be involved, depending on the unique pathophysiology of each disease. Cicatricial pemphigoid, pemphigus vulgaris, and paraneoplastic pemphigus are distinct entities, but all have the potential to have cutaneous and ocular involvement. Awareness and early recognition of ocular involvement in these diseases is important given the increased risk for vision loss and blindness with delay in management...
March 2016: Clinics in Dermatology
Munther M Queisi, Mike Zein, Neerav Lamba, Halea Meese, Charles Stephen Foster
Mucous membrane pemphigoid is a systemic disorder that primarily affects mucous membranes. When localized to the conjunctiva, it is known as ocular cicatricial pemphigoid, a potentially blinding disease. Ocular cicatricial pemphigoid is an indication for systemic immunosuppressive treatment to achieve adequate remission. Immunosuppressive agents are selected with a "stepladder" approach, commencing with medications having the fewest side effects. We provide an update of the literature on immunomodulatory agents since 2011 as additional treatment modalities have been explored in the last 4 years...
May 2016: Survey of Ophthalmology
Noor Tazudeen, Sonoa Au, Jonathan Pewitt, Elmer Tu, Iris K Aronson
A 42-year-old man with ocular erythema and scarring had a conjunctival biopsy revealing depostion of IgM and C3 without IgG at the epithelial basement membrane zone.  Treatment with doxycyline, dapsone, and mycophenolate mofetil was unsuccessful and treatment with rituximab has led to partial remission of the conjunctival inflammation. He has undergone 4 cycles of rituximab treatment at intervals of 12 to 18 months.
October 2015: Dermatology Online Journal
Alessandra Micera, Barbara Stampachiacchiere, Antonio Di Zazzo, Roberto Sgrulletta, Magdalena Cortes, Eduardo Maria Normando, Alessandro Lambiase, Stefano Bonini
OBJECTIVE: In a previous study, we reported the upregulation of Nerve Growth Factor (NGF) and trkANGFR expression in Ocular Cicatricial Pemphigoid (OCP), an inflammatory and remodeling eye disease. Herein, we hypothesize a potential NGF-driven mechanism on fibroblasts (FBs) during OCP remodeling events. To verify, human derived OCP-FBs were isolated and characterized either at baseline or after NGF exposure. MATERIALS AND METHODS: Conjunctival biopsies were obtained from 7 patients having OCP and 6 control subjects (cataract surgery)...
2015: PloS One
Leah L Kammerdiener, Jaime Lynn Speiser, James V Aquavella, Mona Harissi-Dagher, Claes H Dohlman, James Chodosh, Joseph B Ciolino
PURPOSE: To evaluate associations between preoperative diagnosis, soft contact lens (SCL) retention and complications. METHODS: A retrospective chart review was conducted of 92 adult patients (103 eyes) who received a Boston keratoprosthesis type I at the Massachusetts's Eye and Ear Infirmary or the Flaum Eye Institute. Records were reviewed for preoperative diagnosis, SCL retention and subsequent complications. Preoperative categories included 16 autoimmune (Stevens-Johnson syndrome, ocular cicatricial pemphigoid, rheumatoid arthritis and uveitis), 9 chemical injury and 67 'other' (aniridia, postoperative infection, dystrophies, keratopathies) patients...
April 2016: British Journal of Ophthalmology
Philipp Ackermann, Susann Hetz, Julia Dieckow, Martin Schicht, Anja Richter, Charli Kruse, Insa S Schroeder, Matthias Jung, Friedrich P Paulsen
PURPOSE: Aqueous tear deficiency due to lacrimal gland insufficiency is one of the major causes of dry eye disease. In severe cases, such as Sjogren's syndrome, Stevens-Johnson syndrome, or ocular cicatricial pemphigoid, therapy with artificial tears is often insufficient to relieve severe discomfort, prevent progressive ocular surface disease, or enable visual rehabilitation by corneal transplantation. Cell or organ generation from stem cells, resulting in tear-like secretion, presents an option as a suitable alternative treatment...
July 2015: Investigative Ophthalmology & Visual Science
Dandan Zhao, Han Y Yin, Anny Cheng, Rendian Chen, Hosam Sheha, Scheffer C G Tseng
PURPOSE: To report the surgical outcome of "sealing the gap" in treating symblepharon caused by various etiologies other than recurrent pterygium. DESIGN: Retrospective, interventional case series. METHODS: Sixteen eyes of 14 patients with pathogenic symblepharon were consecutively operated by conjunctival recession, sealing the gap between recessed conjunctiva and Tenon capsule with a running 9-0 nylon suture, and covering of the bare sclera with amniotic membrane...
September 2015: American Journal of Ophthalmology
Yakov Goldich, Setareh Ziai, Pichaporn Artornsombudh, Noa Avni-Zauberman, Uri Elbaz, David S Rootman, Clara C Chan
OBJECTIVE: To evaluate the demographic and clinical characteristics of patients referred to a tertiary care hospital cornea clinic for ocular cicatricial pemphigoid (OCP) assessment. DESIGN: Retrospective, nonrandomized, consecutive case series. PARTICIPANTS: Thirty three patients with OCP who were treated at the corneal clinic of Toronto Western Hospital from 2003 to 2012. METHODS: Database search of patients from 2003 to 2012 with a referral request or diagnosis of OCP was conducted at a tertiary care hospital cornea clinic...
April 2015: Canadian Journal of Ophthalmology. Journal Canadien D'ophtalmologie
Mio Yamane, Yoko Ogawa, Masaki Fukui, Mizuka Kamoi, Yumiko Saijo-Ban, Saori Yaguchi, Shin Mukai, Tetsuya Kawakita, Shigeto Simmura, Kazuo Tsubota
PURPOSE: Two new drugs with mucin-inducing and secretion-promotive effects, rebamipide and diquafosol, were recently approved as topical dry-eye treatments. We report two cases in which the long-term use of mucin-inducing eye drops improved chronic ocular graft-versus-host disease (cGVHD)-related dry eye and ocular cicatricial pemphigoid (OCP)-like disease. CASE REPORTS: Case 1. A 61-year-old woman had cGVHD-related dry eye that resisted traditional medications...
April 2015: Optometry and Vision Science: Official Publication of the American Academy of Optometry
C C Drechsler, A Kunze, A Kureshi, G Grobe, S Reichl, G Geerling, J T Daniels, S Schrader
Ocular surface disorders, such as pterygium, cicatricial pemphigoid and external disruptions, can cause severe inflammation, scarring, fornix shortening as well as ankyloblepharon. Current treatments do not resolve these conditions sufficiently. The aim of this study was to evaluate clinical applicability and suitability of plastic compressed collagen to serve as a substrate for the expansion of human conjunctival epithelial cells in order to develop an epithelialized conjunctival substitute for fornix reconstruction...
March 2017: Journal of Tissue Engineering and Regenerative Medicine
Allister Gibbons, Thomas E Johnson, Sara T Wester, Astrid E Gonzalez, Charles C Farias, Carolina Betancurt, Guillermo Amescua, Victor L Perez
PURPOSE: To describe the outcomes and medical management necessary to achieve successful lid surgery in patients with biopsy-confirmed and presumed mucous membrane pemphigoid. DESIGN: Retrospective, interventional case series. METHODS: We included patients with positive biopsy results and cases with a typical clinical active bilateral presentation with negative biopsy results but classic features. We identified 11 operated eyes of 7 patients with lid malposition resulting from mucous membrane pemphigoid, particularly cicatricial entropion, that required surgical correction...
May 2015: American Journal of Ophthalmology
Margaret Zambon, Serban Staicu, Michael Nead, Mark Frampton
Obstructive Airway Disease CasesSESSION TYPE: Affiliate Case Report SlidePRESENTED ON: Sunday, October 26, 2014 at 03:15 PM - 04:15 PMINTRODUCTION: Cicatricial pemphigoid is a chronic inflammatory disease that frequently involves the eyes and upper airway mucosa but very rarely involves the lower airways. We report a patient with CP involving the eyes and sinuses, with worsening obstructive lung disease.CASE PRESENTATION: A 52-year-old man with biopsy-proven CP involving the eyes and paranasal sinuses was referred for pulmonary consultation for poorly controlled asthma...
October 1, 2014: Chest
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