The 2023 iteration of the Global Burden of Diseases, Injuries, and Risk Factors Study (GBD) estimated prevalence, incidence, and health burden for 375 diseases and injuries, including 12 mental disorders. We assess past, current, and emerging trends in the prevalence and burden of mental disorders across sexes and age groups, for 21 regions, 204 countries and territories, and by Socio-demographic Index (SDI) quintile, from 1990 to 2023. Mental disorders included in GBD 2023 were anxiety disorders, major depressive disorder, dysthymia, bipolar disorder, schizophrenia, autism spectrum disorders, conduct disorder, attention-deficit hyperactivity disorder, anorexia nervosa, bulimia nervosa, idiopathic developmental intellectual disability, and a residual category of other mental disorders. A literature review identified epidemiological data for each disorder. These were analysed via a Bayesian meta-regression to estimate prevalence by disorder, sex, age, location, and year. Disorder-specific prevalence was multiplied by disability weights representing the severity of health loss associated with each disorder to estimate years lived with disability (YLDs). Deaths due to anorexia nervosa were assessed with a Cause of Death Ensemble modelling strategy to estimate deaths by sex, age, location, and year, and then multiplied by the standard life expectancy at age of death to estimate years of life lost (YLLs). YLDs equalled disability-adjusted life-years (DALYs) for all mental disorders except anorexia nervosa (the only mental disorder considered as an underlying cause of death in GBD), for which DALYs represented the sum of YLDs and YLLs. We presented prevalence, deaths, YLDs, YLLs, and DALYs as counts, age-specific rates per 100 000 population, and age-standardised rates per 100 000 population. We estimated 1·17 billion (95% uncertainty interval 1·06-1·31) prevalent cases of mental disorders globally in 2023, equivalent to an age-standardised prevalence rate of 14 210·7 cases (12 849·5-15 940·1) per 100 000 population. These estimates represented a 95·5% (75·0-121·2) increase in prevalent cases and 24·2% (11·4-41·4) increase in age-standardised prevalence rate between 1990 and 2023. All mental disorders showed increases in prevalent cases between 1990 and 2023, while notable increases were seen in age-standardised prevalence rates for anxiety disorders, major depressive disorder, dysthymia, anorexia nervosa, bulimia nervosa, schizophrenia, and conduct disorder. There were an estimated 171 million (127-228) DALYs due to mental disorders globally across sex and age in 2023, equivalent to an age-standardised DALY rate of 2070·5 DALYs (1519·1-2750·5) per 100 000 population. Mental disorders contributed to 6·1% (4·8-7·6) of all-cause DALYs in 2023, making them the fifth leading cause of global DALYs (up from 12th in 1990). DALYs were almost entirely composed of YLDs. Mental disorders were the leading cause of YLDs in 2023 (up from second in 1990), explaining 17·3% (14·8-20·6) of all-cause global YLDs. Leading causes of mental disorder DALYs were anxiety disorders (ranked 11th among the 304 diseases and injuries at Level 4 of the GBD cause hierarchy), major depressive disorder (15th), and schizophrenia (41st). Globally in 2023, mental disorder age-standardised DALY rates were higher among females (2239·6 [1643·7-3014·1] per 100 000) than among males (1900·2 [1399·8-2510·8] per 100 000), and peaked in the 15-19 years age group (2617·3 [1850·6-3696·8] per 100 000). All locations showed increased mental disorder DALY rates in 2023 compared with 1990, ranging across countries and territories from 1302·4 (952·7-1683·7) per 100 000 in Viet Nam to 3555·8 (2661·9-4715·0) per 100 000 in the Netherlands. Across SDI quintiles, DALY rates ranged from 1853·0 (1352·1-2469·3) per 100 000 for middle SDI to 2184·1 (1606·1-2890·3) per 100 000 for high SDI. A significant health burden was imposed by mental disorders in all countries and territories in 2023, irrespective of the health resources available. In some instances, this burden has increased over time and is unevenly distributed across populations. Stronger surveillance systems, particularly in low-income and middle-income countries, are required. Additionally, we need more coordinated and inclusive policies to reduce the burden through early treatment and prevention, tailored to sex and age differences across locations. Responding to the mental health needs of our global population, especially those most vulnerable, is an obligation, not a choice. Gates Foundation, Queensland Health, and University of Queensland.
To compare the clinical findings and high-resolution optical coherence tomography (OCT) features of two cases of intraretinal silicone oil (SO) migration in the fovea following repair of fovea sparing rhegmatogenous retinal detachment. In case 1, a 53-year-old female with 20/20 visual acuity had a fovea sparing retinal detachment with grade C proliferative vitreoretinopathy repaired with vitrectomy, scleral buckle, and 1000 centistoke SO tamponade for 5 months. After SO removal, visual acuity was 20/400. In case 2, a 66-year-old-male with 20/25 visual acuity presented with a recurrent fovea sparing retinal detachment repaired with vitrectomy, scleral buckle, and 5000 cSt SO tamponade for 3 months. After SO removal, visual acuity remained 20/25. Intraretinal SO was detected in the fovea on OCT about 4 months after surgery for case 1, and 1 month after surgery for case 2. Intraretinal SO droplets were characterized by focal hyper-reflective light reflexes at the apex and base of SO droplet and increased hyperreflectivity projecting posteriorly. Prototype high resolution (HR) OCT parafoveal layer thickness maps demonstrated ganglion cell layer (GCL) thinning in both cases compared with the contralateral unaffected eye; however, the thinning was more severe in case 1 consistent with worse visual acuity. Over the course of one year HR OCT demonstrated no evidence of adverse reaction to SO and minimal change in SO size and location in both cases. Intraretinal migration of SO into the fovea, after repair of macula sparing retinal detachment, remained stable without evidence of adverse reaction after 1 year of follow-up. Using HR OCT, severe parafoveal GCL thinning, compared to fellow healthy eye, was associated with central vision loss while moderate parafoveal GCL thinning was associated with maintained central vision. Further study is needed to determine if parafoveal GCL thickness measurements can help guide timely SO removal.
Ahmed glaucoma valve (AGV) migration is a rare postoperative complication after AGV implantation. We report a single case of AGV posterior migration in the early postoperative period. A 67-year-old male patient with a history of primary open angle glaucoma, diabetes, and pseudophakia of both eyes presented two days after AGV placement in the right eye. He reported right eye pain, fever, and chills. On examination, he had broken sutures at the valve placement site, purulent discharge from the upper fornix, and an elevated intraocular pressure of 34 mmHg in the right eye. The patient was sent to the hospital due to concerns for orbital cellulitis. Computerized tomography (CT) showed superotemporal post-septal migration of the AGV plate and tube. The patient was admitted to the hospital and started on broad-spectrum intravenous (IV) antibiotics. Cultures of the right eye drainage grew Group A beta-hemolytic streptococci. Six days after the original AGV placement, the patient underwent orbital exploration with removal of the displaced AGV from the posterior third of the globe by an oculoplastics surgeon. The entire AGV plate and tube were removed intact. The wound was irrigated with vancomycin and the conjunctiva was closed. Two days after the AGV removal, the patient showed significant clinical improvement. Upon further investigation, it was determined that the plate was not secured with fibrin glue or suture, only the patch graft was secured with suture. AGV posterior migration following AGV implantation is a rare but serious postoperative complication that must be recognized early and managed appropriately.
To report a case of spontaneous closure of full-thickness macular hole in highly myopic eye. A 68-year-old female with a known history of high myopia presented for routine annual ophthalmologic evaluation. Optical coherence tomography (OCT) was performed revealing an incidental finding of a full-thickness macular hole (FTMH) in the left eye. Given the patient's stable vision and absence of symptoms, a decision was made to observe without surgical intervention. At the 3-month follow-up, repeat OCT demonstrated spontaneous closure of the macular hole with structural and functional improvement of the photoreceptors. Routine imaging in myopic patients can reveal silent but potentially vision-threatening lesions such as macular holes. Although uncommon, spontaneous closure of FTMH in myopic eyes can occur and warrants consideration in patient counselling and management decisions with appropriate caution regarding long-term stability.
To report a case of retinoschisis associated with retinal detachment, documented with ultra-widefield optical coherence tomography (OCT), and to highlight subtle vitreoretinal interface changes not previously described. A patient with retinoschisis was evaluated using multimodal imaging, including ultra-widefield OCT. Serial OCT scans were reviewed to assess structural alterations of the outer and inner retinal layers, vitreoretinal interface, and hyaloid status. Ultra-widefield OCT demonstrated the development of a retinal detachment secondary to an isolated outer retinal hole within the region of retinoschisis, occurring in the context of traction from a thickened inner retinoschisis leaf, without evidence of communication with an inner retinal hole. Additionally, undulations on the inner surface of the outer leaf of the retinoschisis cavity were observed possibly representing residua of a tractional separation between the outer and inner leaf of the retinoschisis cavity. These subtle features were only appreciable through the extended coverage of ultra-widefield OCT imaging. Ultra-widefield OCT provided critical structural insights into the progression from retinoschisis to a retinal detachment. It revealed the development of an isolated outer retinal hole with associated inner leaf traction and undulations on the inner surface of the outer leaf of the retinoschisis cavity without detectable inner leaf holes, underscoring OCT's value in detecting and better understanding subtle vitreoretinal changes in this condition.
Intrastromal misplacement of a foldable intraocular lens (IOL) during cataract surgery is exceedingly rare. Immediate identification of the separation plane is critical to prevent additional corneal injury. A 76-year-old woman with bilateral glaucoma underwent routine phacoemulsification. During insertion of a one-piece acrylic IOL, the lens was inadvertently delivered into the corneal stroma. The IOL remained partially unfolded within a stromal pocket. Intraoperative anterior segment optical coherence tomography (AS-OCT; RESCAN 700) clearly demonstrated deep stromal migration of the IOL and a thick, taut, hyperreflective line consistent with a Type-1 separation of Dua's layer adherent to Descemet's membrane. This real-time confirmation enabled controlled removal of the misdirected IOL without Descemet's membrane rupture, followed by successful in-the-bag implantation of a new IOL and intracameral air tamponade. Postoperative AS-OCT showed gradual resolution of the separation without additional intervention. At 6 months, the cornea remained clear with improved visual acuity (0.6 to 0.7; logMAR 0.22 to 0.15) and an endothelial cell density of 1253 cells/mm2 (52.8% loss). At 17 months, visual acuity remained 0.7, and endothelial cell density was 1158 cells/mm2 (56.4% loss), and corneal clarity was maintained. Intraoperative AS-OCT provided decisive, real-time visualization of the stromal entry plane and Type-1 Dua's layer separation, allowing safe retrieval of the misdirected IOL and preservation of Descemet's membrane integrity. Recognition of this separation pattern is essential for guiding atraumatic management of this extremely rare complication.
To present a proof-of-concept case series documenting the novel in-vivo progression of retinal microaneurysms (MAs) into retinal capillary macroaneurysms (RCMAs) in diabetic retinopathy (DR) using serial optical coherence tomography (OCT). We longitudinally followed four treatment-naïve DR eyes. Over 9 to 30 months, serial OCT clearly captured the enlargement of pre-existing MAs into distinct RCMAs, characterized by a marked increase in wall reflectivity and significant morphological remodelling. This conversion consistently coincided with a notable exacerbation of localized diabetic macular edema (DME) and increased hyperreflective intraretinal dots (HRIDs), indicative of increased inflammation and leakage. The MAs originated in both inner and middle retinal layers. Our series presents the first in vivo, multi-case demonstration of MAs dynamically enlarging into RCMAs, a clinically significant and aggressive form of diabetic microvascular disease. This transformation is consistently associated with worsening CME and increased HRIDs, marking RCMAs as a more exudative and potentially treatment-resistant subset of DME. Emerging evidence, supported by our observations, suggests that many RCMAs may arise at interconnecting vertical channels between the superficial and deep capillary plexuses, where mixed arterial-venous flow and focal pressure gradients predispose to outpouching. These findings challenge current uniform therapeutic approaches, underscoring the importance of serial-OCTs in detecting and characterizing this evolution. Early OCT-based identification could enable more tailored interventions, reducing the risk of severe vision loss. Future research should further clarify systemic and local drivers of this progression, refine OCT biomarkers, and assess whether aggressive or multimodal therapy targeting RCMAs improves patient outcomes.
To report a case of paracentral acute middle maculopathy (PAMM) after physical exertion and to explore possible underlying microvascular mechanisms. A 42-year-old Caucasian man presented with a persistent, unilateral, painless paracentral scotoma that appeared after an intense ski session seven days earlier. His medical history included an 11-year course of migraine with aura and recurrent transient scotomas in either eye, lasting from minutes to days, often triggered by fatigue, exertion, or altitude. At presentation, best-corrected visual acuity was 1.6 Snellen equivalent bilaterally (20/12), with unremarkable slit lamp and fundoscopy examination. Optical coherence tomography (OCT) revealed a focal hyperreflective band in the inner nuclear layer of the right eye, consistent with PAMM. Fundus autofluorescence revealed a hyperautofluorescent spot in the same area. Microperimetry confirmed a localized scotoma in the corresponding area. At follow-up, clinical examination before and after moderate exertion (90 minutes of cycling) showed no new scotomas or clinical abnormalities. However, en face OCT angiography (OCTA) of the deep capillary plexus revealed multifocal variations in capillary density, with some areas demonstrating reduced perfusion after exercise. PAMM is an OCT finding of focal retinal ischemia associated with various vascular disorders. Migraine has also been reported as a risk factor. In this case, physical exertion may have contributed to transient hemodynamic changes, potentially unmasking an underlying retinal microvascular vulnerability. The multifocal post-exercise decrease in perfusion on OCTA despite stable symptoms emphasizes the dynamic, subclinical nature of microvascular injury.Multimodal imaging combining structural and functional assessment is critical for the early detection of ischemic retinal events, even in patients without classic vascular risk factors.
Cutaneous melanoma is the most common malignancy to present with vitreous metastasis. This report describes a case of vitreous metastasis from acral lentiginous melanoma masquerading as intermediate uveitis in a patient undergoing checkpoint inhibitor (CPI) therapy. This is an 86-year-old Caucasian female with Stage III Acral Lentiginous Melanoma of the left heel who was initiated on nivolumab and relatlimab along with intralesional talimogene laherparepvec (TVEC). Eight months later she developed blurry vision and floaters. She was diagnosed elsewhere with unilateral acute intermediate uveitis, leading to discontinuation of immunotherapy and initiation of topical difluprednate. Extensive infectious and autoimmune work-up including MRI of the brain and orbits was negative. Failing to improve, she presented to our hospital. Exam of the right eye showed best corrected visual acuity 20/50, intraocular pressure of 16 mmHg, keratic precipitates, 1+ anterior cells, and 2+ large amelanotic vitreous cells arranged in veils. The left eye was quiet. Vitreous biopsy revealed malignant melanoma cells, confirming metastatic melanoma. The patient underwent a therapeutic pars plana vitrectomy, intravitreal melphalan injection, and ocular radiotherapy, with resolution of vitreous metastasis. Several atypical, red flags in this patient may serve as clinical indicators; history of malignancy with locoregional progression, timing of immune CPI therapy to symptom onset, failure to treatment response, and large white, amelanotic vitreous cells arranged in veils. This case emphasizes the importance of maintaining a broad differential diagnosis in patients with atypical, or treatment resistant uveitis, especially in individuals with known underlying malignancy.
Acetazolamide is the mainstay of treatment for idiopathic intracranial hypertension (IIH). Transient myopia is a rare ocular adverse effect, usually attributed to ciliary body edema and forward displacement of the lens-iris diaphragm. A 44-year-old woman developed IIH during pregnancy following hormonal therapy for in-vitro fertilization. She was started on acetazolamide 1 g/day and, after five doses, experienced sudden bilateral blurred vision that improved with myopic correction and resolved spontaneously within 24 hours after discontinuation of acetazolamide. Examination showed no signs of angle closure or choroidal effusion. Optical coherence tomography (OCT) confirmed preserved ganglion cell layer and no axonal loss. After counselling the patient about the potential for recurrent transient myopia and other acetazolamide-related adverse effects, acetazolamide was reintroduced at a lower dose (500 mg/day). No recurrence of myopia occurred, and papilledema improved markedly over six weeks. This case illustrates a rare idiosyncratic effect of acetazolamide in pregnancy-associated IIH and demonstrates that rechallenge at a lower dose may be tolerated without recurrence.
To report a case of testicular choriocarcinoma with choroidal metastasis in a patient who initially presented with visual symptoms that resulted in the diagnosis and treatment of his underlying malignancy and to summarize prior reports in the literature. Choroidal metastasis is a rare manifestation of choriocarcinoma and reported cases have been associated with substantial vision loss and poor survival. Choroidal metastases from testicular choriocarcinoma are often associated with significant intraocular hemorrhage and can mimic other choroidal lesions such as choroidal melanoma. Multimodal imaging can help differentiate between these lesions. In a patient who presents with a choroidal lesion and hemorrhage, metastatic testicular choriocarcinoma should be considered.
Peripheral nodulocystic corneal degeneration (PNCD) is a rare and poorly understood corneal degeneration. We report a case most consistent with PNCD in a patient with a remote history of conventional laser-assisted in situ keratomileusis (LASIK) and multiple systemic inflammatory conditions. A 52-year-old woman with a medical history of multiple systemic arthralgias and an ocular history of bilateral LASIK presented with progressive left eye dryness and discomfort. Ocular examination revealed multiple bullous corneal nodules in both eyes, predominantly in the superior and temporal periphery of the cornea. Anterior segment optical coherence tomography (AS-OCT) revealed subepithelial cystoid spaces with Bowman's Membrane disruption, and specular microscopy showed mild endothelial cell loss with polymegethism and pleomorphism. The clinical findings were most consistent with peripheral nodulocystic degeneration (PNCD). She declined superficial keratectomy, and her symptoms improved with topical cyclosporine, perfluorohexyloctane, and brimonidine. We describe a case of lucent peripheral corneal bullae most consistent with PNCD, of which only one similar case has been previously reported in the literature. PNCD appears to be an indolent, slowly progressive corneal degeneration. This case highlights a unique presentation of corneal degeneration and cause of peripheral corneal bullae. The interplay between corneal degeneration, systemic inflammatory disease, and prior refractive surgery warrants further investigation.
Corneal allogenic intrastromal ring segments (CAIRS) were developed as a biologic alternative to synthetic intracorneal ring segments and are primarily reported in the management of keratoconus and corneal ectasia. Its role in high corneal astigmatism still limited. A 54-year-old male presented with decreased visual acuity in the left eye due to high regular corneal astigmatism. Preoperative best-corrected visual acuity was 0.8 (decimal scale), with a manifest refraction of: +3.5/9.25 × 109. CAIRS implantation was performed based on the magnitude of astigmatism, and limitations of alternative corrective options. At one year postoperatively, corneal tomography demonstrated a marked reduction in astigmatism with improved corneal regularity. Pachymetry and posterior corneal parameters remained stable, and anterior segment optical coherence tomography (OCT) confirmed appropriate intrastromal segment positioning without complications. This case demonstrates the feasibility of CAIRS implantation in isolated high corneal astigmatism, with stable tomographic outcomes at one year. With careful patient selection, CAIRS may represent a tissue-preserving option in selected cases.
The Smaller-Incision New-Generation Implantable Miniature Telescope (SING IMT) is an IOL designed to enhance central vision in patients with bilateral late-stage AMD. This report describes a case of successful surgical repositioning of a tilted SING IMT following haptic displacement out of the capsular bag. A 77-year-old man with bilateral geographic atrophy and baseline CDVA (Corrected Distance Visual Acuity) of logMAR 0.96 (Snellen 20/200) underwent uncomplicated implantation of a SING IMT in the right eye. Six weeks postoperatively, CDVA showed no improvement, and the patient reported monocular diplopia with off-axis visual perception. Retinal morphology remained stable. Slit-lamp and anterior segment OCT revealed pronounced optic tilt caused by inferior haptic dislocation out of the capsular bag. The haptic was successfully repositioned by performing superior traction with a push-pull hook, resulting in restoration of straight, diplopia-free vision and an improvement in CDVA to logMAR 0.52 (Snellen 20/63). Prompt surgical correction of haptic displacement may fully restore the optical and functional benefits of the SING IMT without compromising corneal health.
This case highlights a rare association between prostaglandin analog therapy and isolated ciliary body effusion. Travoprost is a topical prostaglandin analog frequently prescribed as an intraocular pressure-lowering agent. We report the case of an 84-year-old pseudophakic female with primary open-angle glaucoma who developed ciliary effusion after long-term travoprost use. Patient reported decreased vision in her left eye over two weeks. Ophthalmological examination revealed decreased visual acuity, myopic shift, shallowed anterior chamber, and increased intraocular pressure in the left eye. Fundoscopy was unremarkable. Anterior segment optical coherence tomography revealed presumed ciliary body effusion with anterior displacement of the iris-intraocular lens complex. Significant resolution occurred within one week after discontinuing travoprost. Isolated ciliary effusion should be recognized as a rare adverse effect of travoprost, with drug discontinuation playing both diagnostic and therapeutic roles.
To describe an unusual case of posterior segment inflammation occurring in temporal association with a switch from reference ranibizumab to its biosimilar, Ximluci®. A 77-year-old woman with neovascular age-related macular degeneration presented for an unscheduled visit reporting mild subjective visual loss one day after the second monthly intravitreal injection of ranibizumab biosimilar Ximluci following ten consecutive monthly injections of reference ranibizumab without adverse events. Clinical examination revealed mild optic disc edema, and peripapillary venous leakage on fluorescein angiography, without arteriovenous filling delay or ischemic signs. Multimodal imaging findings were consistent with posterior segment inflammation with venous involvement. Infectious and systemic inflammatory work-up was negative. The patient was treated with systemic and topical corticosteroids, resulting in progressive resolution of inflammatory signs. Due to suboptimal anatomical response of the underlying neovascular lesion and the recent inflammatory episode, therapy was subsequently switched to 2 mg Aflibercept, which was well tolerated without recurrence of intraocular inflammation. This case highlights a rare episode of posterior segment inflammation occurring in temporal association with a switch to a ranibizumab biosimilar. Although ranibizumab biosimilars have demonstrated a favorable safety profile in clinical trials and regulatory assessments, uncommon inflammatory reactions may emerge in real-world settings. Careful clinical monitoring following treatment switching and continued post-marketing surveillance are essential to further characterize the ocular safety of these agents.
We report a unique case of iris metastasis from epidermal growth factor receptor (EGFR)-positive lung adenocarcinoma, diagnosed solely by cytologic analysis of aqueous humor without tissue biopsy. A 56-year-old man with a known history of lung adenocarcinoma presented with unilateral iris masses, anterior chamber inflammation, and secondary glaucoma. Intraocular pressure was initially controlled with cataract surgery combined with microhook trabeculotomy. However, the pressure re-elevated despite maximal topical antiglaucoma therapy (including a prostaglandin analog, beta-blocker, carbonic anhydrase inhibitor, alpha-2 agonist, and Rho-kinase inhibitor) and oral acetazolamide. Three sessions of transscleral cyclophotocoagulation were performed. In addition, two intravitreal injections of aflibercept (2.0 mg each) were administered for rubeosis iridis. Aqueous humor obtained via anterior chamber paracentesis immediately before intravitreal injection demonstrated Class V adenocarcinoma cells on cytology, allowing the diagnosis of iris metastasis without iris biopsy.The lesions subsequently enlarged, and similar iris lesions appeared in the fellow eye during the final 1 month before the patient's death. Systemic disease progression became evident during the final 1 month before death, and the patient ultimately succumbed 9 months after the initial visit. Conclusions and Importance: This is among the few reported cases of iris metastasis confirmed solely by aqueous cytology. This case underscores the diagnostic utility of anterior chamber paracentesis as a less invasive yet reliable alternative to iris biopsy in carefully selected patients. It also highlights the clinical course and therapeutic challenges in managing anterior segment metastasis from epithelial tumors such as lung cancer.
Neuro-ophthalmic manifestations are common in patients with advanced human immunodeficiency virus (HIV) infection and may result from opportunistic intracranial infections. However, partial pupil-sparing third cranial nerve palsy is an uncommon presentation in this population and may be misleading, as it is often presumed to be ischemic. We report an atypical presentation of cerebral toxoplasmosis manifesting as partial pupil-sparing third nerve palsy. We report the case of a 37-year-old HIV-positive male with advanced human immunodeficiency virus infection who presented with headache, seizures, and binocular diplopia and was found to have a partial pupil-sparing third nerve palsy. Neuroimaging revealed multiple intracranial enhancing lesions with surrounding vasogenic edema. Cerebrospinal fluid analysis demonstrated varicella zoster virus positivity, and stereotactic brain biopsy ultimately confirmed cerebral toxoplasmosis. The patient was managed with antimicrobial therapy and showed clinical improvement. In patients with human immunodeficiency virus infection, pupil-sparing third nerve palsy should not be presumed to be exclusively ischemic, as infectious and other opportunistic etiologies may underlie its presentation. Careful clinical assessment, detailed examination, and appropriate neuroimaging are essential for accurate diagnosis and timely management.
Immunoglobulin G4 (IgG4)-related orbital disease is an uncommon fibroinflammatory condition characterized by IgG4-positive plasma cell infiltration of orbital structures. While its features are well-recognized in adults, pediatric cases remain exceedingly rare, with limited reports in the literature. We describe three pediatric female patients with IgG4-related orbital disease, aged 5, 11, and 16 years, who presented with progressive proptosis and globe displacement. Orbital imaging in all cases demonstrated infiltrative lesions involving the lacrimal gland. Histopathologic confirmation was obtained in each, showing dense lymphoplasmacytic infiltrates with cluster of differentiation 138 (CD138) positivity and a high proportion of IgG4-positive plasma cells. Treatment primarily consisted of systemic corticosteroids and immunomodulatory therapy. Rituximab was successfully administered in one refractory case, leading to substantial clinical improvement. Pediatric IgG4-related orbital disease, although rare, should be considered in the differential diagnosis of orbital masses in children, particularly when the lacrimal gland is involved. Histopathology remains essential for diagnosis, and systemic corticosteroids constitute the mainstay of therapy, with immunosuppressants and biologics reserved for refractory disease.
Individuals with hereditary retinoblastoma (RB) carry a heightened risk of developing subsequent malignant neoplasms that is amplified by prior radiation treatment. Here, we present a novel case of intraocular extraskeletal osteosarcoma (ESOS) arising in the phthisical eye of an irradiated, heritable RB survivor. A 23-year-old man with a history of bilateral RB presented with a year of left orbital and eye pain. On examination, the left eye had mucopurulent discharge and exposed orbital tissue. On imaging, the left globe, periocular structures, optic nerve, and optic nerve sheath were surrounded by abnormal contrast enhancement. As a result, the left eye was enucleated. On histopathology, an ESOS filled most of the globe with extraocular extension. The tumor was associated with focal, benign-appearing intraocular bone of the type commonly seen in phthisical eyes. The patient was found to have pulmonary metastases, consistent with metastatic stage IV osteosarcoma. He was started on systemic chemotherapy and underwent an exenteration of the left orbit. This case highlights a rare manifestation of osteosarcoma in an irradiated, heritable RB survivor. It underscores the need for longitudinal ophthalmic examination that potentially includes periodic echography when eyes are phthisical, and oncologic surveillance in RB survivors to facilitate early diagnosis and intervention for life-threatening secondary malignancies. The findings also suggest that osteosarcoma can arise from metaplastic bone within phthisical globes.