Intraepithelial Sebaceous Gland Carcinoma: A Multicenter Retrospective Case Series.

PURPOSE: Intraepithelial sebaceous gland carcinoma is a rare form of sebaceous gland carcinoma, with 10 published case reports to date. The authors report the clinical, histological, and prognostic features of this rare carcinoma. METHODS: This is a multicenter retrospective case series of patients from 3 Australian sites. Information collected included patient demographics, clinical risk factors, initial and subsequent presentations, histology results, management, and outcomes. RESULTS: Twelve cases were identified. The average age of presentation was 72 years (range 52-92 years), with more females (n = 8) affected than males. The most common symptoms and signs were ocular irritation (n = 9) and eyelid mass (n = 8), with a predilection toward upper lid involvement. Five cases of sebaceous gland carcinoma were confirmed on initial histology. Initial management included wide local excision with margin control (n = 11), and primary topical mitomycin C (n = 1). Adjunctive conjunctival mapping biopsy was performed in 5 cases. Recurrence occurred in 7 cases, at an average of 31 months (range 7-83 months) after initial treatment. There were no cases of distant metastasis, however, 2 cases developed local invasion. Management of recurrences included exenteration (n = 2), further excisions alone (n = 3), and excision with adjuvant mitomycin C (n = 2). CONCLUSIONS: Clinicians and pathologists should have a high index of suspicion for primary intraepithelial sebaceous gland carcinoma on the upper eyelid. Close follow-up is recommended, given the high risk of local recurrence.

Clinical and radiological characteristics of odontogenic orbital cellulitis.

PURPOSE: To assess the radiological features and clinical outcomes of odontogenic orbital cellulitis. METHOD: Multi-centre retrospective study of odontogenic orbital cellulitis. Primary outcomes assessed were causal organism(s), clinical signs, radiological findings, management and visual outcomes. RESULTS: Four patients with odontogenic orbital cellulitis were identified for inclusion. There was an equal proportion of men and women with a mean age of 43 years (range 25-56 years). All patients presented with an orbital compartment syndrome, with visual acuity of counting fingers (n = 1, 25%), hand movements (n = 1, 25%) and no perception of light (n = 2, 50%). The organisms implicated were Streptococcus milleri (n = 3, 75%) and Streptococcus constellatus (n = 1, 25%). MRI findings showed a subperiosteal abscess was present in all cases, which was characterised radiologically as a T1-hyperintense, T2 minimally hyperintense collection with restricted diffusion and a low apparent diffusion coefficient signal. Final visual acuity ranged from 6/6 to no light perception. One patient required an orbital exenteration due to extensive necrosis with sepsis and systemic deterioration. CONCLUSIONS: Odontogenic orbital cellulitis carries a serious risk of vision loss with a propensity to present with an orbital compartment syndrome secondary to Streptococcus species. Outcomes were highly variable, with two cases progressing to blindness of which one required an orbital exenteration.

Orbital Myeloma and Plasmacytoma: An Australian Study.

PURPOSE: The purpose of this study was to provide Australian data on the clinical and radiological features and outcomes in patients with orbital plasmacytomas. METHODS: Multicentre retrospective review of orbital plasmacytoma and orbital involvement in multiple myeloma (MM) from 2005 to 2022 in Australia. RESULTS: Twenty-one participants were identified. The median age was 62 years (range 34-88 years), and 11 (52%) were females. Eighteen (84%) had a known diagnosis of MM prior to their orbital presentation, with all patients eventually being diagnosed with systemic MM. Thirteen (72%) were receiving active treatment for systemic myeloma on presentation, while 3 (17%) were in remission. All but 1 had unilateral orbital involvement (n = 20, 95%). Common presenting symptoms and signs were decreased visual acuity (n = 13, 62%), proptosis (n = 11, 52%), limited motility (n = 15, 71%), and optic neuropathy (n = 5, 24%). Radiologically, 15 (71%) involved the superotemporal orbit, 7 (33%) inferotemporal orbit, and 16 (76%) involved ≥1 extraocular muscle. Sixteen (76%) were biopsied and confirmed orbital plasmacytoma on histopathology. Treatment modalities included intravenous and oral steroids (n = 7, 33%), chemotherapy (n = 9, 43%), radiotherapy (n = 13, 62%), stem cell transplant (n = 3, 14%), and surgical debulking and decompression (n = 3, 14%). Mortality was high, with 15 (71%) having MM-related mortality. CONCLUSIONS: This is the largest cohort of Australian data on orbital plasmacytomas. Most patients have a diagnosis of systemic MM at presentation. It is crucial to recognize and treat these patients early due to a poor systemic prognosis.

Differentiation of bacterial orbital cellulitis and diffuse non-specific orbital inflammation on magnetic resonance imaging.

PURPOSE: To examine the radiological differences between bacterial orbital cellulitis (OC) and diffuse non-specific orbital inflammation (DNSOI) on magnetic resonance imaging (MRI). METHODS: Retrospective study of patients with OC and DNSOI with an MRI orbital scan. Localised orbital inflammation (e.g., idiopathic dacryoadenitis and myositis), quiescent orbital inflammation and pre-septal cellulitis were excluded. RESULTS: Thirty-two patients presenting between 2008 and 2023, including twenty-one OC patients (mean age: 42.5 ± 24.9 years old, male: 6), and eleven DNSOI patients (mean age: 52.3 ± 17.8 years old, male: 16). Both OC and DNSOI demonstrate orbital fat contrast-enhancement. However, whilst OC demonstrated a hyperintense T2 signal (P < 0.001), variable signal was observed in DNSOI, with a hypointense T2 signal more suggestive of DNSOI (P = 0.012). When the lacrimal glands were involved, indistinct margins were more likely in OC (P < 0.001), whilst gross enlargement and contrast-enhancement was observed in DNSOI (P = 0.032 and 0.017, respectively). Peripheral contrast-enhancement of the extraocular muscle (EOM) (P = 0.002) was more common in OC, whilst DNSOI demonstrated variable contrast-enhancement throughout the affected EOM (P < 0.001). The presence of contralateral abnormalities, such as lacrimal gland enlargement and EOM involvement, are more suggestive of DNSOI. CONCLUSION: Several MRI features, beyond overt sinogenic disease, may help to differentiate OC from DNSOI, including the orbital fat signal intensity, EOM and/or lacrimal gland involvement, and contralateral orbital abnormalities. However, these features may not be specific, and thus highlights the ongoing radiological dilemma clinicians are faced when tasked with differentiating between infectious and non-infectious orbital inflammatory disease.

Clinico-radiological features of optic nerve sheath schwannoma: Review and illustrative case.

The optic nerve sheath is a rare site for schwannoma, to our knowledge 19 optic nerve sheath schwannoma (ONSS) cases have been reported. Difficulty can arise in diagnosis as imaging findings can be relatively non-specific. We describe a case of ONSS that mimicked orbital cavernous venous malformation pre-operatively. A 43-year-old woman presented with right subacute visual loss, reduced vision to 6/48 and signs of optic neuropathy. Endocapsular excision of the mass, which was adherent to the optic nerve, was accomplished from an endoscopic endonasal approach utilizing a 5-hand technique of retrocaruncular dissection. Histology showed spindle-shaped tumour cells with S100 positivity, consistent with ONSS. At 6 months post-operatively vision had improved to 6/6. We show that an endoscopic endonasal approach, augmented by transcaruncular retraction, can be utilized to excise ONSS with an excellent outcome. We review published cases of ONSS, including demographic and clinical features, differential diagnosis based on radiological features, and described outcomes, which are generally poor.

Myxoma: An Unusual Aggressive Orbital Lesion.

Myxoma is a rare tumor that can be challenging to diagnose, with imaging findings that can be nonspecific. We present a case of a 21-year-old man who presented with a subacute history of right visual deterioration and proptosis. Imaging showed a large right superomedial orbital mass of 43 × 31 × 24 mm, which enhanced heterogeneously and was eroding the adjacent orbital roof and medial wall. An excisional biopsy was performed via a lid crease approach under transorbital endoscopic guidance. We review the clinical, radiological, and histological characteristics of myxoma. In addition, we highlight important associations with genetic syndromes including Carney complex and Mazabraud syndrome.

Non-infectious hypertrophic pachymeningitis associated with orbital inflammatory disease: a pooled analysis.

PURPOSE: To describe four cases of non-infectious hypertrophic pachymeningitis (HP)-associated with orbital inflammatory disease (OID). This study summarises the clinico-radiological features, outcomes, and management of HP-associated OID. METHODS: Retrospective case-series of patients with radiological evidence of HP and OID. Comprehensive literature review of all published English-language non-infectious causes of HP-associated OID. Reference lists were screened for inclusion of relevant articles. RESULTS: Thirty-seven cases of HP-associated OID (Mean age: 49.2 ± 17.4 years old; Male: 15) were identified, including four cases from our institution. Aetiologies included ANCA-associated vasculitis (12/37), non-specific/idiopathic (11/37), IgG4/multifocal fibrosclerosis (11/37), neurosarcoidosis (1/37), inflammatory myofibroblastic tumour (1/37), and giant cell arteritis (1/37). Orbital pain, headache, visual deterioration, and cranial nerve palsies were common clinical presentations. Both "focal" and "diffuse" HP were observed, with the most common sign of orbital involvement being an inflammatory orbital mass, typically with orbital apex involvement. Orbital myositis and dacryoadenitis were less common. The cavernous sinus was the most common site of extra-orbital inflammation. There was no single differentiating specific radiological feature between non-specific and specific forms of HP-associated OID. CONCLUSION: The clinico-radiological manifestations of HP-associated OID differ from those described in isolated HP or OID. There is no single specific radiological marker differentiating non-specific/idiopathic disease from secondary causes; however, the co-existence of HP in OID should prompt suspicion of an underlying cause. The disease may be refractory or resistant to initial treatment, although guidelines surrounding its management and the long-term prognosis remain to be determined.

Microbiology of acute bacterial dacryocystitis: a tertiary institutional experience in South Australia.

PURPOSE: To provide a comprehensive microbiological profile of bacterial dacryocystitis in South Australia. By identifying the specific microorganism and antibiotic susceptibility, this study intends to aid ophthalmologists in choosing appropriate empirical antibiotic therapies and development of evidence-based clinical guidelines. METHOD: A retrospective study was conducted at the Royal Adelaide Hospital (RAH) over five years (2018-2023) of patients with acute dacryocystitis. The study included 43 patients, and data encompassed demographic information, clinical presentation, microbiological analysis, management, and outcomes. Patients with chronic dacryocystitis were excluded. RESULTS: Among the 43 patients included in the study (female 28 (65%), mean age: 64 years old), the most common clinical features were pain (74%) and swelling (70%). Organisms were identified in 49% of patients, with the predominant bacteria being Staphylococcus aureus (42%), Streptococcus species (19%), and Escherichia coli (8%). Aggregatibacter species (8%), Morganella morganii (4%), Enterobacter cloaceae (4%), Hafnia alvei (4%), mixed anaerobes (4%), E coliforms (4%) and Pseudomonas aeruginosa (4%) were also identified. The most frequently prescribed empirical antibiotics were amoxicillin-clavulanic acid (50%), flucloxacillin (33%) and cefalexin (18%). CONCLUSION: The microbiological trends of acute dacryocystitis have largely remained consistent, with a predominance of Gram positive organisms. This is the most recent profile analysis of acute dacryocystitis in South Australia and will help form evidence-based clinical guidelines.

Primary Dacryocystorhinostomy for Acute Dacryocystitis: A Systematic Review.

INTRODUCTION: Acute dacryocystitis is a common condition occurring secondary to nasolacrimal duct obstruction. We aim to assess the efficacy of primary dacryocystorhinostomy for the management of acute dacryocystitis. METHODS: A systematic search of the databases PubMed/MEDLINE, Embase, and CENTRAL was performed to December 2023. Data extraction and risk of bias analysis was conducted in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. RESULTS: Fourteen articles fulfilled inclusion criteria. The results demonstrated that for the treatment of acute dacryocystitis, primary dacryocystorhinostomy (DCR) is anatomically and functionally efficacious, with low complication rates and minimal risk of recurrence. The anatomical success rates for primary endonasal DCR (EnDCR) were 81.8-100%, 83.8-87.5% in delayed EnDCR and 66-100% in delayed external DCR (ExDCR). Functional success was generally defined as a subjective absence of epiphora, which was achieved in 86.4-92.0% of primary EnDCR, and 0-89.5% of the delayed ExDCR cohorts. Two randomized controlled trials established equivalent rates of functional success between primary versus delayed EnDCR (87.5% vs 87.5% and 91.3% vs 92.3%). Increased perioperative bleeding was recorded in 27.8% of primary EnDCR cases in 1 study. ExDCR was infrequently associated with postoperative fistula and scar formation and cicatricial punctal ectropion. The mean length of admission trended to be shorter when the procedure was performed closer to the acute presentation. The admission time for all primary EnDCR cases varied between 1 and 7 days. CONCLUSION: Primary dacryocystorhinostomy is an efficacious and safe management option for acute dacryocystitis. Further analyses of health-economics are required.

Ameloblastic Carcinoma With Orbital Invasion.

Ameloblastic carcinomas are malignant tumors arising from the odontogenic epithelium and defined as having features of ameloblastic differentiation in addition to cytological features of malignancy. Orbital involvement is rare and generally involves invasion of the orbital floor, apex, or soft tissue. This report describes an advanced presentation of ameloblastic carcinoma with orbital invasion and provides a review of the literature. A 58-year-old male presented with a 2-year history of a mid-facial mass, causing intracranial invasion and distortion of most skull foramina, nasopharynx, nasal cavity, and both orbits. Notably, there was an en-plaque pattern of circumferential tracking of the tumor along both orbital walls without invasion beyond the extraconal space, causing compression of the orbital apex and proptosis. Histology demonstrated nests of ameloblastic carcinoma and the advanced tumor was deemed nonresectable, with treatment being palliative.

A Brief History of Lacrimal Bypass Techniques.

PURPOSE: Lacrimal bypass is the creation of a fistula connecting the conjunctiva with the lacrimal sac or nasal cavity. Bypass is indicated in canalicular obstruction or agenesis; sac absence, destruction or prior excision; lacrimal pump failure; or dacryocystorhinostomy failure. We aim to review the various techniques that have been developed over the last century for lacrimal bypass. METHODS: We conducted a comprehensive literature review of techniques which have focused on creating a conduit extending from the conjunctiva or canaliculi to the lacrimal sac, or extending that bypass to the nasal cavity bypass. RESULTS: The main techniques reviewed include canaliculodacryocystorhinostomy, conjunctivodacryocystostomy, conjunctivorhinostomy, conjunctivodacryocystorhinostomy, and conjunctivoductivodacryocystorhinostomy. CONCLUSION: Lacrimal bypass surgery has evolved due to innovation in microsurgical techniques, instruments and materials. Conjunctivodacryocystorhinostomy with Jones tube insertion is the predominant bypass technique, reflecting a culmination of historical developments. Understanding the variety of lacrimal bypass techniques is important for exploring alternative options when necessary.

Qualitative and Quantitative Magnetic Resonance Imaging in Bacterial Orbital Cellulitis.

PURPOSE: To summarise the qualitative and quantitative parameters of bacterial orbital cellulitis (OC) on magnetic resonance imaging (MRI) and explore their clinical correlations. METHODS: Multi-centre retrospective study with inclusion of patients of all ages with OC who underwent MRI. Patients with isolated pre-septal cellulitis, bilateral disease and poor-quality scans were excluded. An enlargement ratio for extraocular muscles (EOMs) was calculated by dividing maximal EOM measurements from the affected side by the contralateral side. RESULTS: Twenty MRI scans from twenty patients (Mean age: 40.8 ± 24.3 years old, M: F = 15:5) between 2011 and 2022 were analysed. Three (15.0%) cases were paediatric patients (<18 years old). All cases had both pre-septal and orbital fat involvement. The EOM were affected in nineteen cases, with the superior muscle complex (18/19, 94.7%) most commonly affected. Mean enlargement ratio (1.30, Range: 1.04-1.82) was greatest for the medial rectus on axial views on T1 and fat-suppressed contrast-enhanced T1 (FS CE T1). Optic peri-neuritis was present in eleven (55.0%) patients, whilst two (9.5%) cases had optic neuritis. A greater degree of proptosis was observed in patients with optic neuropathy and those who underwent surgical intervention compared to those without (p = .002 and p = .002, respectively). CONCLUSION: MRI remains an important imaging modality for evaluating complicated OC. However, qualitative features may lack accuracy and is not a reproducible means of analysis. Simple quantitative parameters, such as proptosis and EOM measurements, correlate with high-risk clinical features and may have utility in predicting clinical course.

Corneal Neurotization for Neurotrophic Keratopathy: A Multicenter Experience.

PURPOSE: Corneal neurotization is an emerging technique that offers potential for visual rehabilitation in neurotrophic keratopathy. This study reports on a multicenter experience and outcomes for both direct and indirect methods of corneal neurotization. METHODS: Retrospective case series. Sixteen patients with neurotrophic keratopathy who underwent corneal neurotization across 5 centers in Australia and Israel were identified for inclusion. Corneal neurotization was performed via direct neurotization from the ipsilateral or contralateral supraorbital/supratrochlear nerve or by the use of an interpositional sural nerve graft. Change in corneal sensitivity (measured in millimeters by the Cochet-Bonnet aesthesiometer), visual acuity, and corneal health. RESULTS: Over a mean follow-up period of 31.3 months (range: 3 months-8 years), mean corneal sensitivity improved from 3.6 mm (range: 0-25 mm) to 25.3 mm (range: 0-57 mm). Visual acuity improved on average from 20/380 to 20/260. Twelve of 16 patients (75.0%) improved in at least 2 out of the 3 main outcome measures. Nine patients (56.3%) showed an improvement in visual acuity; 13 (81.3%) showed an improvement in average corneal sensitivity; and 11 (68.8%) showed an improvement in corneal health. There were no intraoperative or postoperative complications. CONCLUSIONS: Corneal neurotization is an emerging surgical treatment option for the management of neurotrophic keratopathy. With appropriate case selection, outcomes are favorable and complication rates are low, for a condition that is otherwise challenging to manage. Patients with severe neurotrophic keratopathy should be considered for this surgical treatment option.

Corrigendum: Smartphone-delivered multicomponent lifestyle medicine intervention for improving mental health in a nonclinical population: a randomized controlled trial.

[This corrects the article DOI: 10.3389/fpubh.2023.1231981.].

Magnetic Resonance Imaging of Idiopathic Orbital Myositis.

PURPOSE: To characterize the qualitative and quantitative features of idiopathic orbital myositis (IOM) on MRI. METHODS: This was a multicenter retrospective study of patients with active IOM with MRI. Patients with incomplete clinical records, poor-quality or interval scans without active myositis, and specific orbital myositis (i.e., orbital myositis secondary to an identified condition) were excluded. An enlargement ratio was calculated by dividing the diameters of the affected extraocular muscle (EOM) by the contralateral unaffected EOM. RESULTS: Twenty-four patients (mean age: 44.4 ± 17.8 years-old, male: 11) between 2011 and 2022 were included. One case (4.2%) was pediatric (17 years old), and 6 cases presented with recurrence. Active IOM was characterized by fusiform EOM enlargement, high T2 signal, and contrast enhancement. Average maximal EOM diameters ranged from 4.6 to 7.7 mm (enlargement ratio: 1.4-2.2). Eighteen (75%) patients had single EOM involvement, most commonly the medial rectus. Other ipsilateral structures affected included focal orbital fat (16/24, 66.7%) and lacrimal gland (8/24, 33.3%). Contralateral changes in the EOM and/or lacrimal gland were observed in 7 patients (29.2%). Patients presenting with recurrence were likely to develop ongoing recurrent episodes ( p = 0.003). CONCLUSIONS: Various radiological patterns of involvement described including EOM enlargement, contrast enhancement, abnormal signal, and involvement of other orbital structures are indicative of active IOM. IOM remains a heterogeneous spectrum of acute and chronic clinico-radiological presentations. Inflammation may involve other ipsilateral or contralateral orbital structures or may be bilateral despite presenting clinically as unilateral disease. Quantitative measurements may have utility in differentiating IOM from other causes of orbital myositis.

Medication-associated orbital inflammation: A systematic review.

We performed a comprehensive systematic review to identify medication-associated orbital inflammation and to characterize its clinico-radiological features. We reviewed English-language articles describing medication-associated orbital inflammation (i.e., orbital myositis, dacryoadenitis and orbital fat) published to June, 2023. Isolated inflammation of the intraocular structures or globe alone (i.e. uveitis, scleritis, optic neuritis and perineuritis) were excluded. In medication-associated orbital inflammation, the extraocular muscles are preferentially affected, occurring in isolation or in combination with other orbital and/or intraocular structures. Clinico-radiological manifestations may be non-specific; however, certain medications may be distinguished according to the presence of systemic prodrome, laterality, associated intraocular inflammation, and predisposition to involve certain orbital structures. Rapid identification, discontinuation of the provoking medication, and systemic corticosteroid therapy (if appropriate) typically achieves a favorable visual prognosis. As new medications become adopted by clinicians, rare adverse effects will be further delineated.Medication-associated orbital inflammation is an important diagnostic consideration in orbital inflammatory disease. A careful medication history and clinical assessment may be revealing, permitting timely discontinuation of the offending agent and initiation of appropriate management.

Recurrent inferior oblique myositis and its outcomes.

This report presents a unique case of recurrent idiopathic inferior oblique myositis (IOM) with a focus on clinico-radiological characteristics and histological features. A woman in her early 40s presented with a third episode of IOM following a 12-year period of quiescence. The first two episodes were characterised by unilateral IOM with rapid resolution following oral prednisone treatment. MRI revealed anterior focal enlargement of the left inferior oblique muscle with ipsilateral lacrimal gland enlargement. An inferior oblique muscle and lacrimal gland biopsy demonstrated significant inflammatory infiltrate. An intraorbital injection of triamcinolone acetonide was administered with complete resolution of symptoms within 1 week.

IgG4-related Orbital Disease Presenting as an Orbital Apex Lesion.

IgG4-related disease is an uncommon multisystem inflammatory disease characterized by tissue infiltration by IgG4 plasma cells, extensive fibrosis, and sclerosing inflammation. Diffuse orbital involvement extending to the orbital apex and cavernous sinus has been reported, but a solitary well-circumscribed lesion at the apex has not been previously reported. Herein, we report a unique case of IgG4-related orbital disease presenting as a solitary well-circumscribed orbital apex lesion causing subacute visual decline in a 45-year-old male. MRI demonstrated bilateral lacrimal gland and infraorbital nerve enlargement and an 18 × 7 mm left orbital apex lesion demonstrating homogeneous contrast enhancement and isointense signal on T2. He was initially treated with tapering systemic corticosteroid therapy with improvement in his vision; however, he experienced visual deterioration 3 months later with recurrent inflammation. He subsequently commenced high-dose systemic corticosteroid therapy and rituximab infusions with improvement of left visual acuity to 6/7.5 + 2.

Sinonasal Chondrosarcoma With Unusual Orbital Invasion.

A case is presented of a 43-year-old male with a chronic history of progressive nasal obstruction and epiphora. MRI confirmed a heterogeneous mass involving the middle and superior turbinates with T2 hyperintense and calcified components, with extension into the inferomedial orbit. Tissue biopsy revealed a grade 2 chondrosarcoma of the conventional subtype. Endonasal wide local resection of the lesion was performed with clear margins. The patient had no functional sequelae and will undergo routine surveillance.

Inferior orbital fissure release to access the inferolateral orbital apex.

OBJECTIVE: To describe release of the inferior orbital fissure (IOF) as a novel surgical technique that will improve access to the inferior and inferolateral orbital apex. DESIGN: Laboratory investigation. PARTICIPANTS: Human cadaver heads. METHODS: Cadaveric dissection study whereby the technique of an IOF release was performed. A swinging eyelid preseptal approach was used to enable a subperiosteal dissection to the orbital floor without obstruction from orbital fat prolapse. A plane was dissected between periorbita and Muller's muscle, the smooth muscle that overlies the entire length of the IOF. Measurements were taken in the sagittal plane from a point on the inferior orbital rim directly above the infraorbital foramen to the most posterior aspect of the orbital floor. RESULTS: Fifteen orbits from 8 cadaver heads were dissected with the aid of a rigid 0-degree endoscope and microsurgical instruments. This study demonstrated that an additional 10.93 ± 2.10 mm of access to the orbital apex was gained after the release of periorbita from the IOF. CONCLUSION: We propose that the IOF release can be adopted as an adjunctive technique to orbital surgery of the inferior and inferolateral orbital apex.