Morbihan Disease: A Rare Case of Periorbital Bilateral Edema with Histopathological Findings of Chronic Inflammation and Demodex Localization.

BACKGROUND Morbihan disease, also known as Morbihan syndrome, is a rare medical condition characterized by chronic facial edema predominantly affecting the upper two-thirds of the face. Despite being recognized in medical literature for decades, its true prevalence and underlying pathophysiology remain poorly understood. Various hypotheses, including impaired lymphatic drainage, abnormal vascular permeability, immune dysregulation, and inflammatory reactions to demodex infestation, have been proposed to explain the etiology. CASE REPORT We present a case of a 61-year-old man with organized periocular edema of the upper third of the face, ultimately leading to Morbihan disease diagnosis. The patient underwent a midface lift, allowing for tissue retrieval for histopathological examination of the eyelid edematous skin, which revealed chronic inflammation, ectasia of small lymphatic vessels, and features of demodex intrafollicular localization. These findings were not specific, but consistent with the diagnostic hypothesis. The patient was referred to a rheumatologist for further evaluation and treatment. He did not respond well to systemic corticosteroids and immunosuppressive therapy. Rather, this resulted in extension of the edema to the upper eyelid. The patient opted not to undergo further treatment. CONCLUSIONS Morbihan disease is often misdiagnosed due to its rarity and overlapping clinical features with other facial conditions. Its management is challenging and can require a combination of medical and surgical interventions. Systemic corticosteroids, immunosuppressive agents, and topical treatments have had varying success. Surgical procedures, such as blepharoplasty or laser therapy, can be considered in severe cases. Early recognition and appropriate management are crucial to improving patient outcomes and quality of life.

"Demodicosis" Mimicking PreSeptal Cellulitis: Severe Periocular and Facial Inflammation Caused by "Normal" Skin Flora.

Due to their relatively high prevalence and commensalism, the pathogenicity of Demodex mites has been debated. Recent data, however, show Demodex to be associated with skin and ocular surface diseases such as rosacea, blepharitis, and keratitis. Here the authors report the first known case, to the best of the their knowledge, of Demodex infestation mimicking preseptal cellulitis in an adult human. A 29-year-old male bilaterally blind from advanced retinopathy of prematurity presented with a 2-month history of right-greater-than-left upper eyelid and periocular/cheek swelling, redness, and ocular discharge that did not resolve with oral antibiotics or oral steroids. Based on MRI findings, biopsies of the right lacrimal gland, right orbital fat, and right upper eyelid preseptal skin were obtained which revealed marked intrafollicular Demodex mite density and budding yeasts in the upper eyelid skin. This case serves to alert clinicians to this entity that may not otherwise be usually considered in ophthalmic clinical practice.

Case Report: Ophthalmomyiasis Externa Caused by Cordylobia anthropophaga Masquerading as a Lid Abscess.

Myiasis is a neglected tropical disease caused by the larvae of dipterous flies. Cutaneous infestation is the predominant type documented in sub-Saharan Africa, and ocular involvement is uncommon. We report the rare occurrence of a case of eyelid ophthalmomyiasis caused by Cordylobia anthropophaga in a Nigerian female to raise awareness among practitioners in both tropical and nonendemic areas.

The effect of Blephadex™ Eyelid Wipes on Demodex mites, ocular microbiota, bacterial lipase and comfort: a pilot study.

PURPOSE: To investigate the effect of Blephadex™ Eyelid Wipes on Demodex mites, ocular microbiota, bacterial lipase, tear film characteristics and ocular comfort after one month of daily use. METHODS: Twenty subjects were randomly assigned to use the Blephadex™ Eyelid Wipes on either eye once daily for 30 days whilst the contralateral eye was left untreated in this observer-masked, within-subject study. Demodex count, eyelid bacterial colony count, Tearscope Plus non-invasive tear break up time (NITBUT), Lipiview® tear film lipid layer thickness and phenol red thread test tear volume were measured at baseline and 30 days. Bacterial lipase was quantified from single bacterial colonies using a glycerol monolaurate assay. Ocular comfort was assessed at both visits using the Ocular Surface Disease Index (OSDI) questionnaire and visual analogue scales (VAS) to capture monocular symptoms of itching, dryness and overall discomfort. RESULTS: Six males and 14 females, median age 63.5 (range 48-76) completed the study. A statistically significant reduction in Demodex count was observed in treated eyes only (median ±â€¯IQR: treated eyes 2 ±â€¯3 vs. 0 ±â€¯2, ANOVA p = 0.04). Bacterial colony count, lipase production, NITBUT, lipid layer thickness and tear volume remained unchanged (p > 0.05). Overall comfort improved over time in treated eyes only (15 ±â€¯32 vs. 10 ±â€¯16, p = 0.05). Dryness symptoms significantly reduced in both treated and untreated eyes (23 ±â€¯42 vs. 12 ±â€¯21 and 23 ±â€¯41 vs. 10 ±â€¯15, p = 0.02). The OSDI and ocular itch scores remained unchanged (p > 0.05). CONCLUSION: In this pilot study, no changes were observed in ocular microbiota, tear film characteristics or bacterial lipase in eyes treated with Blephadex™ Eyelid Wipes after one month of daily use in this normal healthy population. Although a statistically significant reduction in Demodex count was observed in treated eyes, overall numbers of Demodex were low. A parallel group, placebo-controlled, randomised clinical trial in a population with active blepharitis is warranted to further elucidate these preliminary findings.

The correlation between the microstructure of meibomian glands and ocular Demodex infestation: A retrospective case-control study in a Chinese population.

Meibomian gland dysfunction (MGD) is a common disease in ophthalmic clinic. This study aimed to explore ocular Demodex infestation on the microstructure changes of the meibomian glands (MGs) in patients with MGD by in vivo confocal microscopy (IVCM).We retrospectively reviewed 103 eyes of 52 patients with MGD and 62 eyes of 31 non-MGD patients. All enrolled patients underwent IVCM examination. The following IVCM parameters were recorded: the MG acinar density (MAD), MG acinar longest diameter (MALD), MG acinar shortest diameter (MASD), MG orifice area (MOA), severity of MG fibrosis (MF), MG acinar irregularity (MAI), meibum secretion reflectivity (MSR), inhomogeneous appearance of walls of acinar units (AWI) and periglandular interstices of acinar units (API), and the number of Demodex.The positive rate of Demodex infestation in MGDs was 89.32%, and statistically higher than control group (controls; P < .001). All parameters showed statistically significant differences between MGDs and controls (P < .001), and Demodex-negative group and Demodex-positive group (P < .05) in both MGDs and controls, except MAD (P = .826) in controls. The number of Demodex was positively correlated with MALD, MASD, MF, MAI, MSR, AWI, and API in MGDs and controls (P < .05), and negatively correlated with MAD and MOA in MGDs (P < .05). MOA showed a strong significant correlation with the number of Demodex in controls (P < .001), whereas there was no significant difference between the number of Demodex and the MAD in controls (P = .448).Demodex can cause microstructural changes of MGs, which can cause or aggravate MGD, and the more the number of Demodex infestation, the more serious the structural damage.

Evaluation of the ocular surface characteristics and Demodex infestation in paediatric and adult blepharokeratoconjunctivitis.

BACKGROUND: To evaluate the ocular surface characteristics and the infestation of Demodex in Chinese paediatric and adult blepharokeratoconjunctivitis (BKC). METHODS: Fifty consecutive patients with BKC and 50 age- and sex-matched healthy subjects were enrolled. Lid margin characteristics and corneal disorders were evaluated under slit-lamp illumination. Four eyelashes were collected from each eye to examine Demodex infestation by light microscopy. RESULTS: Corneal neovascularization (P = 0.001) and scarring (P = 0.040) were significantly worse in children than in adults with BKC, whereas meibum quality was worse in adults (P = 0.008). Diagnosis delay was longer in children with BKC than in adults (2.2 vs 1.2 years, P = 0.022). Demodex infestation was more frequent in subjects with BKC than in healthy subjects (56% vs 26%, P = 0.002). The lid margin inflammation and meibomian gland dysfunction were worse in Demodex-positive subjects than in Demodex-negative subjects with BKC. CONCLUSIONS: Children with BKC had severer corneal disorders compared with adult BKC patients, which may be caused by a long-delayed diagnosis. Ocular demodicosis was more common in subjects with BKC. Ocular Demodex infestation was associated with worse lid margin inflammation and meibomian gland dysfunction.

Periocular thelaziasis presenting as an orbital mass - a case report.

Thelazia callipaeda is a rare parasitic infestation caused by spiruroid nematode of the genus Thelazia. We report a case of a 74-year-old gentleman who presented with a painless swelling of left lower lid since 15 days. Examination revealed a firm mobile mass along the inferior orbital rim. Magnetic Resonance Imaging showed a well-defined preseptal cystic lesion and Ultrasound screening revealed multiple mobile worms within. Patient underwent cyst excision in toto under local anesthesia. Four long refractile worms were isolated from within the cyst cavity. Species identification confirmed the parasite as Thelazia callipaeda. Periocular thelaziasis usually presents as free floating worms in the conjunctival sac, anterior chamber or vitreous cavity. It is important to be aware of this rare entity which should be considered as a differential diagnosis in endemic areas.

Case series: periocular habronemiasis in five horses in the Netherlands.

In tropical and subtropical climates, infection of periocular tissue by Habronema larvae is a recognised cause of conjunctivitis or blepharitis. To the authors' knowledge, only a few cases of habronemiasis have been described in Western Europe, and it has not been documented previously in the Netherlands. The objective of this report is to describe the occurrence of five cases of (peri)ocular habronemiasis in the Netherlands, of which four date from the past few years. The diagnosis was based on the history, clinical signs and histopathologic examination of biopsy specimens. A granulomatous conjunctivitis/dermatitis and sulphur-like granules were present in all cases. Histopathology showed an eosinophilic granulomatous inflammation, and three out of five (60 per cent) samples revealed one or more nematodes on section. Treatment combinations with surgical excision, local corticosteroid and/or anthelmintic drugs were used. Furthermore, all horses received ivermectin or moxidectin. Treatment resulted in healing of the lesions in four horses. One case, which was refractory to treatment, resolved spontaneously after the onset of colder weather. This case series suggests an increased prevalence of (peri)ocular habronemiasis in the Netherlands. This diagnosis should therefore be considered when being presented with a horse with granulomatous conjunctivitis/dermatitis in Western Europe, especially during the summer months.

External ophthalmomyiasis due to Dermatobia hominis. A case report. / Oftalmomiasis externa por Dermatobia hominis. A propósito de un caso.

CLINICAL CASE: A 46-year-old woman with no relevant medical history, native of Honduras and resident in Spain for one and a half months. The patient went to the Emergency Department due to inflammation of the upper eyelid of the right eye, with an area that simulated an abscess. This was drained (obtaining hardly any purulent content). Treatment was prescribed with oral and topical antibiotics, as well as an anti-inflammatory drug. One week later the patient returned, with improvement of the inflammatory signs, but with discomfort and corneal erosions. After eversion of the upper eyelid, a «worm¼ type parasite emerged from the tarsus. The extraction was completed with a clamp, and was later identified as Dermatobia hominis (Dh) by examination of a fresh specimen. The subsequent outcome of the patient was favourable. DISCUSSION: Preseptal cellulitis in patients from tropical and sub-tropical areas can be caused by Dh.

[Tick Infestation of the Eyelid: Case Report and Literature Review]. / Parasitose Palpebral por Carraça: Caso Clínico e Revisão da Literatura.

Tick infestation is rarely seen in ophthalmological practice. We report the case of a 73-year-old woman who presented with edema and erythema of her right upper eyelid. Biomicroscopic examination revealed a tick firmly attached to the eyelid margin. The tick was successfully removed using fine tweezers, and within a few days, the patient had fully recovered without sequelae. Ticks are a threat to public health as they are known vectors of numerous infectious agents responsible for serious pathologies such as Lyme disease. Ticks may also induce cutaneous local reactions, and more rarely, tick paralysis. Since morbidity increases substantially with the duration of the infestation, the tick should be completely removed as soon as possible, and patients should be aware about possible signs of tick related diseases.

No âmbito da prática clínica oftalmológica, as parasitoses por carraças são incomuns. Os autores descrevem o caso clínico de uma doente de 73 anos observada por edema e eritema da pálpebra superior direita. O exame oftalmológico revelou uma carraça aderente à margem palpebral. A carraça foi removida usando uma pinça fina, e poucos dias após, houve regressão total dos sinais inflamatórios, sem registo de complicações. O parasitismo por carraças representa uma ameaça para a Saúde Pública, pois as carraças são vetores de inúmeros agentes infeciosos responsáveis por patologias potencialmente graves, como a doença de Lyme. As carraças podem também induzir reações cutâneas locais e, mais raramente, paralisia neuromuscular. A morbilidade associada à picada da carraça aumenta com a duração da parasitação, pelo que a carraça deve ser removida o mais rápido possível, e os doentes devem ser alertados para os sinais de possíveis complicações locais e sistémicas.