Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery最新文献

Necrobiotic xanthogranuloma (NXG) is a rare systemic disease, that commonly manifests with orbital and ocular adnexal involvement, presenting with periocular yellow papules and plaques that may ulcerate. Periorbital skin lesions are a hallmark of the disease, and in their absence the diagnosis may be delayed, preventing prompt systemic evaluation and appropriate treatment of this condition. We report a unique case of a 58-year-old female patient with NXG that presented with severe bilateral necrotizing scleritis, left orbital mass and no cutaneous manifestations of NXG. This case highlights the importance of considering NXG, despite absence of skin lesions, when other ophthalmological manifestations are present. Recognition of NXG is crucial due to the high lifelong risk of developing hematological malignancy.

Immune checkpoints refer to mechanisms entrusted with the modulation of immune responses in peripheral tissues and are required for minimising collateral damage. Immune checkpoint inhibitors (ICPi) work through numerous pathways, including the anti-CTLA-4 (cytotoxic T-lymphocyte-associated protein 4), anti-PD-1 (programmed cell death protein 1) and the PD-L1 (protein cell death protein-ligand-1) pathways. They are proving to be an exciting therapeutic avenue in the attempt to activate anti-tumour activity. Ipilimumab is a fully human monoclonal antibody working on the anti-CTLA-4 pathway, while nivolumab and pembrolizumab are humanised monoclonal IgG4 antibodies that work on the PD-1 pathway. Despite a growing body of research pertinent to these novel therapies, early indications show that they are limited by their side effect profile. Furthermore, their efficacy appears to be greater in cancers with a high mutational burden. We present two female patients with bilateral reactive dacryoadenitis secondary to ICPi therapy, a finding that to the best of our knowledge was not previously described in the literature.

Sebaceous carcinoma is a known mimicker of benign conditions, leading to frequent delays in diagnosis and proper treatment. We present two patients with chronic cicatrizing conjunctivitis initially diagnosed as ocular mucous membrane pemphigoid (MMP) and later found to have sebaceous carcinoma. Both patients presented with unilateral conjunctivitis that failed to improve with topical and systemic therapy, eventually developing fornix foreshortening and extensive symblepharon. Case 1 was diagnosed with ocular MMP based on clinical features alone, while Case 2 was diagnosed with biopsy-negative disease. Months to years later, both patients developed lid lesions found to be sebaceous carcinoma and underwent exenteration. As diagnosis and treatment of ocular MMP without positive direct immunofluorescence testing becomes increasingly accepted, clinicians should consider sebaceous carcinoma as the initial diagnosis or as a developing phenomenon during immunosuppression in the setting of chronic inflammation. A low threshold for repeat biopsy should be maintained.

Purpose: In oculoplastic surgery the eyelid tissue is frequently stretched in order to repair defects after tumor surgery. However, there is a paucity of research regarding how stretching affects eyelids. The purpose of this study was to gain insight into how traction force affects eyelid stretch as well as tissue perfusion, using a laser-based in vivo monitoring technique.

Method: Lower-lid pentagonal resections were performed in eight patients and a total of nine eyelids. The medial section of the eyelid was then stretched using a dynamometer up to a force of 2.3 Newtons (N), and eyelid stretching and blood perfusion were continuously measured using laser speckle contrast imaging.

Results: Tissue perfusion decreased exponentially when eyelid tissue was stretched, with an initial sharp decline followed by a more gradual reduction. Perfusion approached zero at a force of approximately 2.0 N. The length of the eyelid increased with increasing force up to 1.5 N, after which there was only a very slight increase in length.

Conclusions: Eyelid tissue seems to respond to traction in a non-linear fashion, where the initial force results in the greatest eyelid stretching and reduction in blood perfusion. The results provide information on the effects of a large force for direct closure of large eyelid defects. Considering how quickly perfusion approaches zero, the high success rate of eyelid reconstruction surgery is likely a testament to the extensive vascularization of the periocular region.

Intraorbital lymphatic-venous malformations are rare lesions that represent a therapeutic challenge given their location and high rate of recurrence, with only a few cases in adult patients having been published in the literature. We present the case of a 30-year-old male with a right intraorbital lymphatic-venous malformation treated with sirolimus at a dose of 4 mg/day with complete clinical and radiologic remission. Mild cold-like symptoms ensued during the first week of treatment and elevation of liver function enzymes and D-dimer occurred in the context of acute SARS-CoV-2 pneumonia. No major adverse effects were documented. After 18 months of treatment, the patient remains asymptomatic and ophthalmologic examinations including optical coherence tomography and visual field test are within normal values.

Purpose: The gold standard for skin cancer diagnosis is surgical excisional biopsy and histopathological examination. Several non-invasive diagnostic techniques exist, although they have not yet translated into clinical use. This is a proof-of-concept study to assess the possibility of imaging an angiosarcoma in the periocular area.

Methods: We use laser speckle, hyperspectral, and photoacoustic imaging to monitor blood perfusion and oxygen saturation, as well as the molecular composition of the tissue. The information obtained from each imaging modality was combined in order to yield a more comprehensive picture of the function, as well as molecular composition of a rapidly growing cutaneous angiosarcoma in the periocular area.

Results: We found an increase in perfusion coupled with a reduction in oxygen saturation in the angiosarcoma. We could also extract the molecular composition of the angiosarcoma at a depth, depicting both the oxygen saturation and highlighting the presence of connective tissue via collagen.

Conclusions: We demonstrate the different physiological parameters that can be obtained with the different techniques and how these can be combined to provide detailed 3D maps of the functional and molecular properties of tumors useful in preoperative assessment.

Mucous membrane graft (MMG) is used for moderate-to-severe cicatricial entropion repair either in primary or recurrent cases. We conducted a review to comprehensively summarize the various surgical techniques, outcomes, and complications of using MMG for cicatricial entropion. Though the comparison between different techniques is limited by multiple factors like small numbers of cicatricial entgropion patients, variable severity and success criteria across studies, and different underlying cicatricial entropion etiologies, the author has brought forth the nuances of the use of MMG for cicatricial entropion repair along with its outcomes and complications. MMG use in moderate-to-severe cicatricial entropion gives favourable outcomes. The shortened tarsoconjunctiva is lengthened using MMG, which is used either with terminal tarsal rotation or anterior lamellar recession (ALR) or tarsotomy alone. Non-trachomatous entropion has poor outcomes compared to trachomatous entropion. The most common source of MMG is labial or buccal mucosa and the exact size of MMG harvested is variable according to the defect, and very few prefer oversizing the graft by 10-30%. The outcomes of ALR+MMG appear similar to tarsal rotation and MMG for severe cicatricial entropion. The recurrences of trichiasis or entropion can occur for up to one year after surgery, irrespective of the technique used. Factors affecting the outcomes of cicatricial entropion repair are not well known. There is a non-uniformity in data reporting across literature; hence, future studies with details on severity of entropion, ocular surface changes, forniceal depth and ocular surface inflammation, and the degree of dry eye disease would be informative.

A 42-year-old non-menopausal, non-pregnant woman presented with an incidentally noted right orbital apex lesion producing mild compressive optic neuropathy. Imaging revealed an apical, intraconal, inhomogenously enhancing mass consistent with a cavernous venous malformation displacing the optic nerve. The patient was monitored over a 15-year period with serial orbital imaging and clinical examinations and ultimately demonstrated significant regression in the size of the lesion, accompanied by the complete resolution of optic neuropathy. She was non-menopausal when the regression was noted and remained non-pregnant throughout the follow-up period.

Purpose: To evaluate the results of a minimally invasive combined endoscopic and eyelid crease/medial suprabrow incision approach in collaboration with oculoplastic and sinus surgeons for the treatment of recurrent/recalcitrant sino-orbital mucoceles.

Methods: Eighteen cases of recurrent/recalcitrant sino-orbital mucoceles, treated in collaboration with oculoplastic and sinus surgeons at the University of Michigan, were retrospectively reviewed. The recurrence of mucocele, reduction in proptosis, and complications were evaluated.

Results: The mean age at the time of surgery was 49 years (range: 17-76 years). All cases had a history of previous sinus or orbital surgeries for mucoceles. Among 18 cases, eight were due to chronic sinus infections, six due to trauma, three due to Schneiderian papilloma, and one case was secondary to an inflammatory sinus disease. Thirteen cases (72%) presented with orbital or facial cellulitis, while five cases (38%) experienced periocular swelling and limited extraocular motility. Following a mean follow-up of 19 months (range: 1-76 months)), recurrence was observed in two cases (11%): one in a cystic fibrosis patient with chronic sinusitis, and the other in a case of Schneiderian papilloma. The mean pre-operative proptosis in the affected eye was 2.78 mm, with an average decrease of 2.33 mm after surgery. Complications occurred in two cases, including one case of hypoesthesia in the forehead and one case of post-operative strabismus.

Conclusion: Our series of 18 cases of recurrent/recalcitrant mucoceles, with only two cases of recurrence, demonstrates that this minimally invasive approach can be successfully employed for advanced sino-orbital disease, with a low rate of adverse outcomes and aesthetically pleasing results.

A 75-year-old immunocompetent male presented with a right orbital cellulitis after a foreign body penetrating injury. He was taken for orbitotomy with foreign body removal and started on broad-spectrum antibiotics. Intra-operative cultures were positive for Cladophialophora bantiana, a mold known for causing brain abscesses with no prior reports of orbital invasion in the literature. Following culture results, the patient was managed with voriconazole and required multiple orbitotomies and washouts for infection control.