Guardians of the ocular surface: lessons learned from a challenging case of Langerhans cell histiocytosis with eyelid involvement.

Langerhans cell histiocytosis comprises a heterogeneous range of clinical manifestations secondary to clonal proliferation of histiocytes, characterized by the accumulation of these cells in various organs and tissues. The ophthalmological component commonly involved is the orbit. Herein, we report a rare case of Langerhans cell histiocytosis with eyelid involvement, which resulted in severe ocular surface complications, which subsequently significantly impacted the patient's quality of life. This case report highlights the fact that despite being rare, Langerhans cell histiocytosis should be included in the differential diagnosis of eyelid lesions. Furthermore, a multidisciplinary approach with a systemic overview is crucial for managing the ocular complications.

Granulomatosis with Polyangiitis Discovered Because of Repeated Upper Eyelid Swelling.

Background and objectives: The initial symptom that triggers granulomatosis with polyangiitis (GPA) diagnosis is rarely ocular. We describe a case with a single ocular lesion identified as probable GPA due to proteinase 3 (PR3)-antineutrophil cytoplasmic antibody (ANCA)-positivity according to the diagnostic criteria of the Ministry of Health in Japan; the lesion repeatedly worsened. Materials and methods: A 25-year-old female visited the Department of Ophthalmology, Asahi General Hospital, with upper eyelid swelling and conjunctival and episcleral hyperemia of the left eye. Both hordeolum and eyelid cellulitis were suspected, as the condition was resistant to treatment with antibiotic eye drops. Episcleritis was suspected due to localized hyperemia in the upper part of the eye. Upon treatment with antibacterial agents and steroid eye drops, the swelling and the hyperemia repeatedly worsened every week. Results: Blood samples were positive for PR3-ANCA, and GPA with an isolated ocular lesion was considered. After oral steroid treatment, the patient had no recurrence for 4 years. There was no systemic involvement in the upper respiratory tract, lungs, or kidneys. Conclusions: Diagnosing GPA with ocular symptoms as initial manifestations is challenging. GPA should be considered in treatment-resistant eyelid, orbital, and episcleral lesions, even at a young age.

Eyelid dermatitis in patch-tested adult patients: a systematic review with a meta-analysis.

Eyelid dermatitis (ED) affects a cosmetically significant area and leads to patients' distress. Despite ongoing and recent research efforts, ED remains a multidisciplinary problem that needs further characterization. We aimed to evaluate the atopic eyelid dermatitis (AED) frequency in ED patients and to perform their clinical profiling. PubMed databases were searched from 01.01.1980 till 01.02.2024 to PRISMA guidelines using a search strategy: (eyelid OR periorbital OR periocular) AND (dermatitis or eczema). Studies with patch-tested ED patients were included. Proportional meta-analysis was performed using JBI SUMARI software. We included 65 studies across Europe, North America, Asia and Australia, with a total of 21,793 patch-tested ED patients. AED was reported in 27.5% (95% CI 0.177, 0.384) of patch-tested ED patients. Isolated ED was noted in 51.6% (95% CI 0.408, 0.623) of 8453 ED patients with reported lesion distribution, including 430 patients with isolated AED. Our meta-analysis demonstrated that the AED frequency in patch-tested ED patients exceeded the previous estimate of 10%. Isolated AED was noted in adult patients, attending contact allergy clinics. Future studies are needed to elucidate the global prevalence and natural history of isolated AED in adults.

[A case of eyelid fistula after cosmetic lateral canthoplasty].

A 35-year-old female presented with a chief complaint of exudates from the outer corner of the left eye for more than half a year after cosmetic lateral canthoplasty. A fistula was seen in the skin of the left eye 5 mm from the lateral canthus, with clear fluid inside it. Left eyelid fistula was diagnosed and surgically removed. The histopathological examination confirmed that the tissue connected with the fistula was lacrimal gland tissue. No recurrence was found during the 2-month follow-up.

A rare case of trigeminal trophic syndrome with periorbital cellulitis and full-thickness upper eyelid defect in an undiagnosed patient with human immunodeficiency virus: a case report.

BACKGROUND: Trigeminal trophic syndrome is a rare cranial and facial condition caused by damage to the central or peripheral branches of the trigeminal nerve. This syndrome consists of a triad of anesthesia, paresthesia, and crescent-shaped facial ulcer involving the ala nasi and sometimes extending to the upper lip. Although previous screening for human immunodeficiency virus in some patients with trigeminal trophic syndrome was negative, we present a unique case of trigeminal trophic syndrome who tested positive for human immunodeficiency virus with eye complications. CASE PRESENTATION: We present a rare case of trigeminal trophic syndrome in a 44-year-old Black African woman who tested positive for human immunodeficiency virus. She presented with a 6-week history of progressive, persistent, and painless left sided facial and scalp ulcerations that started as small skin erosion. Diagnosis of trigeminal trophic syndrome was made on clinical grounds based on the triad of anesthesia, paresthesia, and unilateral crescent-shaped ulcer in the trigeminal dermatome and her past medical history. The ulcer healed completely after counseling and pharmacological therapy, but she later developed left periorbital cellulitis and left upper eyelid full-thickness defect. CONCLUSION: This is by far the first documented case of trigeminal trophic syndrome with a positive human immunodeficiency virus test. Testing for human immunodeficiency virus in patients with trigeminal trophic syndrome is necessary as this can help improve clinical management and treatment outcomes. Seeking the services of specialists remotely in resource constraint settings is beneficial for managing complications associated with trigeminal trophic syndrome.

Effects of Preoperative Intravenous Versus Subcutaneous Tranexamic Acid on Postoperative Periorbital Ecchymosis and Edema Following Upper Eyelid Blepharoplasty: A Prospective, Randomized, Double-Blinded, Placebo-Controlled, Comparative Study.

PURPOSE: To compare the effects of preoperative tranexamic acid (TXA) administered intravenously (IV) versus subcutaneously on postoperative ecchymosis and edema in patients undergoing bilateral upper eyelid blepharoplasty. METHODS: A prospective, double-blinded, placebo-controlled study of patients undergoing bilateral upper eyelid blepharoplasty at a single-center. Eligible participants were randomized to preoperatively receive either (1) 1 g of TXA in 100 ml normal saline IV, (2) 50 µl/ml of TXA in local anesthesia, or (3) no TXA. Primary outcomes included ecchymosis and edema at postoperative day 1 (POD1) and 7 (POD7). Secondary outcomes included operative time, pain, time until resuming activities of daily living, patient satisfaction, and adverse events. RESULTS: By comparison (IV TXA vs. local subcutaneous TXA vs. no TXA), ecchymosis scores were significantly lower on POD1 (1.31 vs. 1.56 vs. 2.09, p = 0.02) and on POD7 (0.51 vs. 0.66 vs. 0.98, p = 0.04) among those that received TXA. By comparison (IV TXA vs. local subcutaneous TXA vs. no TXA), significant reductions in edema scores occurred in those that received TXA on POD1 (1.59 vs. 1.43 vs. 1.91, p = 0.005) and on POD7 (0.85 vs. 0.60 vs. 0.99, p = 0.04). By comparison (IV TXA vs. local subcutaneous TXA vs. no TXA) patients treated with intravenous and local subcutaneous TXA preoperatively were more likely to experience shorter operative times (10.8 vs. 11.8 vs. 12.9 minutes, p = 0.01), reduced time to resuming activities of daily livings (1.6 vs. 1.6 vs. 2.3 days, p < 0.0001), and higher satisfaction scores at POD1 (8.8 vs. 8.7 vs. 7.9, p = 0.0002). No adverse events occurred were reported. CONCLUSION: In an analysis of 106 patients, preoperative TXA administered either IV or subcutaneously safely reduced postoperative ecchymosis and edema in patients undergoing upper eyelid blepharoplasty. While statistical superiority between intravenous versus local subcutaneous TXA treatment was not definitively identified, our results suggest clinical superiority with IV dosing.

Scleral Contact Lens to Preserve a Corneal Graft in Chronic Lagophthalmos.

Facial burns involving the periorbital region may lead to cicatricial ectropion and lagophthalmos, causing severe exposure keratopathy and eventually blindness if uncorrected. In these patients, it is critical to provide aesthetic and functional surgical correction to protect the ocular surface from chronic desiccation in addition to visual rehabilitation. Conventional methods may not be sufficient to provide visual rehabilitation in complex cases. Scleral lenses can be a multipurpose alternative for these patients. Herein, we present the challenging case of a patient who developed cicatricial lagophthalmos and exposure keratopathy after facial transplantation due to gasoline burns and received a scleral contact lens for visual rehabilitation.

Burn-Induced Cicatricial Eyelid Retraction: A Challenging Case and Review of Management Principles.

Thermal and chemical burns can result in cicatricial eyelid retraction, characterized by an abnormal resting position of the eyelid margin and increased palpebral fissure height. Eyelid retraction often leads to exposure keratopathy, which can cause complications ranging from mild dry eye to globe-threatening ulceration and perforation. Prompt intervention includes aggressive lubrication, moisture chambers, eyelid tarsorrhaphy, and retraction repair surgery. Discussed here is a burn patient that developed severe cicatricial eyelid retraction and ectropion, leading to severe exposure keratopathy and infectious corneal ulceration with perforation. The patient required aggressive medical intervention, as well as 2 surgeries to restore the normal eyelid anatomy to protect the globe.

Contact allergens responsible for eyelid dermatitis in adults.

Allergic contact dermatitis has been established as the most frequent cause of eyelid dermatitis, but it is often misdiagnosed. The purpose of this study was to evaluate the characteristics of patients with eyelid dermatitis who were referred for patch testing. The patients were divided into three subgroups in this retrospective study: patients with only eyelid involvement, patients with involvement of eyelids and other areas, and patients without eyelid involvement. Data was collected on diagnoses, medical history, personal care products and make-up use, occupational dermatitis, and positive allergens. An independent t-test, one-way ANOVA, and chi-squared test were used to analyze the data. A total of 427 patients who referred for patch tests were included in the study. Of these, 139 patients had eyelid dermatitis. Allergic contact dermatitis (ACD) was the most common diagnosis in all three groups referred for patch tests. Use of shaving cream and hair conditioner was significantly higher in patients with only eyelid involvement and nickel sulfate was the most common allergen among them. Patch testing is the gold standard tool in the evaluation of eyelid contact dermatitis, and it is a necessity in the treatment of eyelid dermatitis, for the accurate identification of responsible allergens.

Cerclage sling technique using temporalis muscle fascia to manage paralytic lagophthalmos: A preliminary study.

PURPOSE: Despite various existing surgical techniques, treatment of facial nerve palsy remains difficult. The purpose of this report is to present the cerclage sling technique using temporalis fascia to manage paralytic lagophthalmos. METHODS: A series of six patients underwent a cerclage sling technique using temporalis muscle fascia to treat paralytic lagophthalmos. The technique is presented in detail. Symptoms, palpebral fissures, and lagophthalmos were assessed pre- and postoperatively. Data were submitted for statistical analysis. RESULTS: After surgery, all patients achieved a reduction in clinical symptoms. The upper eyelids had lowered, and the inferior eyelids had elevated, reducing ocular exposure even if mild residual lagophthalmos was present. CONCLUSION: Cerclage using the temporalis muscle fascia sling technique is a safe and effective procedure to treat facial nerve paralytic lagophthalmos. A reduction in ocular exposure and lagophthalmos provides improvement in clinical symptoms and eyelid function.

Outcome Comparison of Transconjunctival Müller's Muscle Recession With Levator Disinsertion (TMRLD) to Gold Weight Implantation in the Treatment of Paralytic Lagophthalmos.

PURPOSE: To compare clinical outcomes of transconjunctival Müller's muscle recession with levator disinsertion (TMRLD) to the traditional gold weight implantation in patients with paralytic lagophthalmos. METHODS: A retrospective nonrandomized comparative review of patients who had gold weight implantation and TMRLD surgeries for paralytic lagophthalmos from January 2016 to January 2023 was performed. The main outcome comparisons were measurement changes in lagophthalmos, marginal reflex distance 1, visual acuity, and corneal examination. Complication and reoperation rates were also compared. RESULTS: Twenty-six cases of gold weight implantation and 20 cases of TMRLD surgeries were identified. The changes in logMAR visual acuity between gold weight implantation and TMRLD groups were not statistically significant (-0.10 ± 0.48 vs. +0.05 ± 0.14, p > 0.05). The percent improvement in lagophthalmos (62.2% ± 51.8% vs. 58.4% ± 21.1%) and final marginal reflex distance 1 (2.22 ± 1.42 vs. 2.25 ± 1.41 mm) were also comparable between groups ( p > 0.05). Both groups showed similar changes in marginal reflex distance 1 (1.75 ± 1.31 vs. 2.83 ± 1.37 mm) and lagophthalmos (3.77 ± 3.92 vs. 3.36 ± 1.36 mm) ( p > 0.05). The overall complication (15.4% vs. 15.0%) and reoperation rates (15.4% vs. 15.0%) were comparable over the follow-up duration (291.6 ± 437.3 vs. 121.0 ± 177.8 days) ( p > 0.05). CONCLUSION: TMRLD is as safe and effective as the gold weight implantation in addressing paralytic lagophthalmos in patients with facial nerve palsy.

Congenital Eyelid Imbrication and Floppy Eyelid Syndromes Complicated by Eversion due to Prostaglandin E Infusion in a Patient With Trisomy 21 and Tetralogy of Fallot.

Congenital eyelid imbrication syndrome is a rare eyelid finding where a long upper lid overlaps the lower lid when the eyes are closed. To date, congenital eyelid imbrication syndrome has been described in the literature less than 10 times. We present a case of congenital eyelid imbrication syndrome in a patient with trisomy 21 and tetralogy of Fallot on a prostaglandin E infusion to maintain a patent ductus arteriosus prior to definitive heart surgery. While on the infusion, the patient developed peripheral edema and flushing due to vasodilation. This coincided with eyelid swelling, conjunctival chemosis, and eversion of the eyelids. Upon cessation of the prostaglandin E1 infusion, his eyelid eversion resolved.

Tarsal Buckling and Entropion after Levator Aponeurosis Advancement in Patients with Floppy Eyelid Syndrome.

Two patients with floppy eyelid syndrome presented with severe eye pain and foreign body sensation after recent levator aponeurosis advancement. The examination in both patients was notable for entropion of the upper eyelid, and upper lid eversion revealed deformity of the tarsus in both patients. Surgical revision with full-thickness horizontal tarsotomy and limited excision of the abnormal tarsus corrected the deformity. The authors propose a mechanism for this phenomenon and a modification of surgical technique that may prevent this complication.

Surgical Correction of Cicatricial Lower Eyelid Retraction: A Systematic Review.

BACKGROUND: Cicatricial lower eyelid retraction is a challenging condition. It involves scarring of the lower eyelid, which causes it to retract and expose the sclera. This can lead to complications such as dry eye syndrome and corneal melting. It can be caused by trauma, burns, or previous eyelid surgery. Detailed assessment and understanding of eyelid anatomy and retraction are critical for successful surgical planning. Dynamic and static examinations of the eyelid including measurements of the lower eyelid margin reflex distance (MRD2) and scleral show are also essential to determine the appropriate treatment approach. METHODS: A systematic review was conducted using Medline, Scopus, and Cochrane databases with keywords related to cicatricial lower eyelid retraction. The publication language was limited to English after 2000. A total of 29 articles were included for data extraction and analysis. RESULTS: The main surgical techniques include tarsoconjunctival grafts, spacers, midface lift, and lateral canthal tendon suspension, although no single procedure has been universally recognized as the gold standard. New innovations such as synthetic grafts and xenografts are being explored for their potential in eyelid reconstruction. Severe cases, defined as those with inferior scleral show greater than 2 mm, may require a combination of reconstruction methods. CONCLUSIONS: Correcting cicatricial lower eyelid retraction is a major challenge in oculoplastic reconstruction. The surgical approach should be individualized, considering the pathologies and etiologies of lid retraction. In-depth knowledge and careful surgical planning are essential for best outcomes. There is no gold standard technique, and postoperative outcomes, complications, and management vary depending on the surgical approach used.