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Purpose: To describe the rare entity of concurrent herpetic acute retinal necrosis (ARN) and orbital inflammation. Observations: Two cases of ARN with simultaneous orbital inflammation are described. A 40-year old male presented with a painful left eye and hand motion visual acuity (VA). Both panuveitis and orbital inflammation with involvement of the sclera and optic nerve were observed. He was initially treated with oral steroid therapy, after which the orbital inflammation improved but the panuveitis remained. A diagnostic anterior chamber paracentesis was positive for HSV-2 by PCR. He was started on systemic antivirals, but ultimately developed a retinal detachment. The second patient was an 18-year old female with hand motion VA in the left eye. Panuveitis and severe conjunctival chemosis were observed. MRI demonstrated dacryoadenitis with preseptal inflammation. The patient was initially started on oral steroid therapy, which alleviated the orbital inflammation but not the intraocular inflammation. An anterior chamber diagnostic paracentesis was positive for HSV-1, after which the patient underwent vitrectomy for a retinal detachment. The patient was started on systemic antiviral therapy. Conclusions and Importance: Herpetic disease should remain on the differential for cases of concurrent intraocular and orbital inflammation. Early recognition of this process may help prevent severe vision loss. It is important to recognize that orbital inflammation secondary to herpetic disease may be diverse in its presentation. Keywords: Acute retinal necrosis (ARN), Orbital inflammation

Purpose To describe a case of Cytomegalovirus (CMV) Retinitis in an immunocompetent patient following Dexamethasone Intravitreal Implant (DII). Observations Retrospective chart review of a single patient. An 80-year-old immunocompetent male presented with floaters in his left eye 10 weeks after his DII. He was noted to have a visual acuity of 20/1200 in his left eye and a panuveitis with CMV retinitis. The patient underwent a vitreous biopsy and given immediate intravitreal foscarnet and an ongoing management regimen of oral valganciclovir for two months. His vision improved to 20/200 and his CMV retinitis resolved. Conclusion and Importance DII can trigger CMV retinitis in immunocompetent patients.

Purpose To report a case of panuveitis and retinal vasculitis associated with pembrolizumab therapy for metastatic lung cancer. Observations A 71-year-old man, who was diagnosed with metastatic lung cancer (squamous cell carcinoma), presented with blurry vision 2 weeks after the initiation of pembrolizumab monotherapy. His best-corrected visual acuity (BCVA) was 20/20 OU, and slitlamp examination revealed fine keratic precipitates, anterior chamber cells (1+) and flare (1+) in both eyes. Dilated fundus examination showed no remarkable finding in the right eye and vitreous haze (2+), perivascular exudates, and vessel sheathing in the left eye. Fluorescence angiography demonstrated dye leakage from the optic disc and both retinal arteries and veins extending from the posterior to the peripheral retina in both eyes. The patient was diagnosed with panuveitis and retinal vasculitis as Grade 3 immune-related adverse event (irAE). Pembrolizumab was discontinued and oral prednisone 70mg/day was given for 1 week. The dose was reduced to 30mg/day for the next 3 weeks and was then stopped. One month after the treatment, intraocular inflammation became quiescent. With a good response to the treatment of irAE, pembrolizumab was restarted. Recurrence of ocular inflammation occurred over the next 1.5 years, but all of which were successfully treated with sub-Tenon's injection of triamcinolone acetonide (STTA). The patient maintained BCVA of 30/20 OU at the latest visit. Conclusions and importance We showed a case of retinal vasculitis occurred as an irAE of pembrolizumab for metastatic lung cancer. Retinal vasculitis was well managed with transient pembrolizumab discontinuation and oral corticosteroid therapy, and pembrolizumab was restarted with the aid of STTA. As ocular irAEs might be controlled by local corticosteroid therapy, the decision to continue immune checkpoint inhibitor therapy should be made on a case-by-case basis.

Purpose To report a case of presumed ocular sarcoidosis initially presenting with features of multiple evanescent white dot syndrome (MEWDS) with atypical optical coherence tomography angiography (OCTA) findings. Observations A 23 year-old woman presented with a unilateral central scotoma, photophobia, and decreased visual acuity after a viral illness. Examination of the right eye revealed multiple round white macular spots and stippled granularity at the fovea. Multimodal imaging with fluorescein angiography (FA), indocyanine green angiography (ICG), fundus autofluorescence (FAF), and optical coherence tomography (OCT) was consistent with a diagnosis of MEWDS. However, OCTA demonstrated choriocapillaris (CC) flow deficits, which is not typical for MEWDS. The clinical course was initially consistent with MEWDS, with spontaneous recovery of symptoms over ensuing months. The patient presented five months later with floaters and a central scotoma. Examination showed panuveitis, and systemic evaluation revealed an elevated angiotensin converting enzyme (ACE) and hilar lymphadenopathy on chest x-ray consistent with presumed sarcoidosis. Conclusions and Importance A case of MEWDS atypically demonstrated CC flow deficits on OCTA and subsequently presented as uveitis secondary to presumed sarcoidosis. Atypical features in MEWDS may be a sign of another disorder masquerading early on as MEWDS and ought to prompt further investigation.

Purpose To report a case of intravitreal methotrexate treatment and fluocinolone acetonide (Retisert®) implantation in a patient with Vogt-Koyanagi-Harada syndrome (VKH). Observations A 34-year-old male was referred for worsening vision and bilateral panuveitis consistent with VKH. He was treated with prednisone, mycophenolate mofetil, prednisolone acetate eye drops, and injections of triamcinolone and adalimumab. He failed to improve with these therapies and developed multiple adverse effects, including hepatotoxicity, severe eye pain, cataracts, and cystoid macular edema. We treated him with intravitreal methotrexate injections in both eyes, which rapidly improved his eye pain, inflammation, and vision. He subsequently underwent fluocinolone acetonide (Retisert®) implantation, cataract extraction with intraocular lens insertion, and Ahmed tube placement for long-term intraocular pressure control. His vision improved from hand motions to 20/30, intraocular pressure remained stable at 17, there was complete resolution of his panuveitis and uveitic macular edema, and his systemic medications were able to be discontinued. Conclusions /Importance: This case demonstrates intravitreal methotrexate may successfully treat intraocular inflammation, pain, and macular edema in VKH. Excellent long-term vision and reduction of adverse effects of systemic medications were also achieved with subsequent fluocinolone acetonide implantation. Combining these two targeted therapies may be an effective strategy in treating VKH in patients who have severe pain and cannot tolerate systemic therapy.

Purpose Toxoplasma gondii is the most common cause of infectious posterior uveitis worldwide in immunocompetent patients. Despite its prevalence, diagnosis can still be challenging and vision-threatening in cases with atypical presentations. This case exemplifies the importance of clinical exam and additional workup when required despite negative initial serology results. Observations A 73-year-old immunocompetent woman presented with a 2-year history of recurrent panuveitis and retinal necrosis not responsive to systemic antiviral therapy. Toxoplasma serum antibodies (IgG and IgM) were not detected on systemic workup one year prior. The slit-lamp exam revealed mutton fat keratic precipitates, panuveitis, and necrotic retinal lesions adjacent to a retinal scar. Repeated Toxoplasma serum antibodies (IgG and IgM) were again negative. However, aqueous fluid testing by polymerase chain reaction (PCR) was highly positive for Toxoplasma gondii. The patient improved after starting systemic anti-toxoplasma therapy. Conclusion/Importance To our knowledge, this is the first report in the literature of an immunocompetent patient with ocular toxoplasmosis and undetectable serum IgG and IgM. Aqueous fluid PCR testing is useful in suspected ocular toxoplasmosis in patients with vision-threatening lesions despite negative serology., Highlights • This is the first report in the English literature of seronegative ocular toxoplasmosis in an immunocompetent patient. • Diagnosis of ocular toxoplasmosis can be challenging. • Clinical exam and additional workup still have a role in excluding ocular toxoplasmosis in immunocompetent patients. • Treatment is recommended in vision-threatening infections.

Purpose To report a case of transient anisocoria and mydriasis in a 14 year old boy with toxoplasmosis retinochoroiditis. Observation The patient presented with panuveitis and mydriasis which persisted for 18 days and spontaneously resolved. Conclusions and importance Mydriasis is a rare potential neurological manifestation of toxoplasmosis retinochoroiditis. Clinicians should be aware of this rare cause of anisocoria.

Purpose To describe an unusual case of bilateral acute retinal necrosis (ARN) that was caused by varicella zoster virus in one eye and Epstein-Barr virus in the fellow eye. Observations A 67-year-old immunocompromised man presented with ARN in the left eye following a dermatomal vesicular rash, with an aqueous sample positive for varicella zoster virus. Four months later, the patient presented with panuveitis and serous retinal detachment in the right eye, with vitreous sample positive for Epstein-Barr virus and negative for varicella zoster, herpes simplex, and cytomegalovirus. Conclusions and importance We report a rare case of bilateral ARN with independent infection of each eye by different viruses; varicella zoster in the left eye and, four months later, Epstein-Barr virus in the right eye. Immunocompromised patients are vulnerable to ARN from any of its inciting viral causes, and intraocular fluid should be obtained for diagnostic confirmation from the second eye in cases of bilateral ARN.

Purpose: To report two rare cases of chronic myeloid leukemia (CML) on tyrosine kinase inhibitors presenting as bilateral serous retinal detachment and ocular inflammation, simulating Vogt-Koyanagi-Harada (VKH) disease. Methods: Case series and review of literature. Result: Two young patients (one male and one female) with CML on treatment with tyrosine kinase inhibitors (imatinib and dasatanib) under remission presented with bilateral sudden vision loss. One patient had bilateral multiple pockets of serous retinal fluid while the other had panuveitis with exudative retinal detachment. There was neither prodromal symptoms nor systemic signs and symptoms suggestive of VKH in both cases. They responded well to systemic steroid therapy without recurrences with complete visual recovery. Conclusion and importance: CML patients can have features similar to VKH even during stable hematological phase and may be possibly associated with the use of tyrosine kinase inhibitors. Hence it is important not to misdiagnose and treat such patients with long term immunomodulators. Keywords: Chronic myeloid leukemia, Panuveitis, Vogt-Koyanagi-Harada disease, Tyrosine kinase inhibitors, Dasatinib, Imatinib

Purpose To report the spectral domain optical coherence tomography (SD-OCT) features of a focal retinitis in an elderly male patient with bilateral syphilitic panuveitis. Observations In the left eye (LE), spectral domain SD-OCT images during the active period revealed hyperreflectivity extending through the full thickness of the retina with no individualization of the layers, except for the retinal pigment epithelium. Once the lesion healed, SD-OCT imaging revealed an inner retinal atrophy and a mild disruption of the retinal pigment epithelium. Conclusions and importance In our patient, treponemal infection seemed to produce full-thickness retinal damage with partial involvement of the retinal pigment epithelium. The severe retinal damage, in this case, led to a poorer visual outcome than in other forms of syphilitic retinal involvement.

Purpose: To describe an atypical presentation of Tubulointerstitial Nephritis and Uveitis (TINU), with challenges in treatment course. Observations: A 12-year-old Hispanic female presented to the National Eye Institute's Uveitis clinic with bilateral blurred vision, red eyes and photophobia, not responsive to topical steroids. On exam, she had bilateral severe panuveitis with areas of subretinal fluid. During her evaluation, she was noted to have elevated serum creatinine. A kidney biopsy confirmed the presence of severe tubulointerstitial nephritis and interstitial fibrosis. She was treated with oral steroids with excellent resolution of symptoms and subretinal fluid. She continued to have anterior segment flares with attempts to taper oral prednisone which lead to treatment with multiple immunomodulatory agents. Associated hypertension and kidney damage complicated the choice of a secondary immunosuppressive agent. Conclusions and Importance: Although rare, TINU can present as panuveitis with choroidal involvement which may or may not be preceded by tubulointerstitial nephritis. A renal biopsy is required for definitive diagnosis, but abnormal urinalysis or renal function should raise suspicion for TINU. Keywords: Immunosuppression, Inflammation, Tubulointerstitial nephritis, Uveitis

Purpose: To report two cases of retinal vasculitis associated with CREST syndrome, a novel ocular finding. Observations: We report two cases of patients with CREST syndrome with ocular inflammatory disease. Patient 1 presented with a right unilateral panuveitis with extensive retinal vasculitis and evidence of prior uveitis in the contralateral eye. Patient 2 presented with a left branch retinal artery occlusion and bilateral retinal vasculitis. Both patients underwent treatment with prednisone and mycophenolate motefil. Conclusions and importance: Retinal vasculitis has not been previously reported in CREST syndrome. Prompt therapy with immunomodulatory therapy can potentially minimize ocular morbidity. Keywords: CREST syndrome, Retinal vasculitis

Purpose To report a case of profound hypotony maculopathy as a complication of single-agent glaucoma therapy in a patient with a history of panuveitis without previous filtering surgery. Observations A 70-year old Hispanic male with a history of resolved bilateral panuveitis, chronic angle closure glaucoma, and pars plana vitrectomy was started on topical timolol 0.5% daily in the left eye for mildly elevated intraocular pressure (15 mmHg). The patient returned 1.5 weeks later with new onset hypotony (1 mmHg), chorioretinal folds, and cystoid macular edema in the same eye without associated signs of inflammation. The drop was discontinued. The patient returned 1 month later with normalized eye pressure and improved vision with near-resolution of chorioretinal changes on optical coherence tomography (OCT). Conclusions and importance Hypotony maculopathy is most commonly seen following glaucoma filtering surgery and ocular trauma. The development of hypotony maculopathy following the administration of topical glaucoma medication alone is rare. Our case is the first to our knowledge to describe the rapid onset of visually significant hypotony maculopathy characterized by profound OCT changes upon the administration of a single topical glaucoma agent in a patient without prior filtering surgery. Treatment with glaucoma medications in patients with complex ocular histories including uveitis and vitreoretinal surgery requires caution and close follow-up.

Purpose: Relapsing polychondritis is a rare multiorgan disease characterized by repeated episodes of inflammation and deterioration of cartilages. We report a case of relapsing polychondritis that presented with severe panuveitis. Observations: A 53-year-old man visited our hospital because of ocular pain in both eyes of 2 weeks' duration. His best-corrected visual acuity was 20/20 in both eyes but he had severe hyperemia of the conjunctiva bilaterally. Inflammation in the anterior segment and vitreous opacity had been getting worse in his right eye. Systemic and topical treatments were not effective, and the visual acuity of the right eye was reduced to hand motion. Thus, pars plana vitrectomy with silicone oil tamponade was performed. After the operation, the vitreous opacities and white lesions in the retina were completely resolved. His visual acuity was improved to 20/20.Three years later, he developed dizziness and swelling of both auricles of his ears, and he was found to have sensorineural deafness. He was diagnosed with relapsing polychondritis after a laryngoscopic examination.Twelve months after the diagnosis, scleritis and panuveitis developed in his left eye, and his visual acuity fell to 20/2000. We performed pars plana vitrectomy with silicone oil tamponade on his left eye. After the vitrectomy, the inflammation of the left eye was resolved. Conclusion: and importance: Ophthalmologist should be aware that severe panuveitis with vitreous opacities may be the initial signs of relapsing polychondritis. In addition, vitrectomy was effective for the treatment of the ocular complications. Keywords: Relapsing polychondritis, Panuveitis, Type II collagen, Pars plana vitrectomy

Purpose: To report two cases of panuveitis in immunocompetent patients in which cytomegalovirus was involved. Observation: Case 1 was a 46-year-old man who had a history of recurrent anterior chamber inflammations in his left eye. After Nd:YAG laser posterior capsulotomy, he developed panuveitis with vitreous haze and periphlebitis. Polymerase chain reaction (PCR) examination revealed the presence of cytomegalovirus (CMV) DNA in the anterior chamber (AC). He responded well to a series of intravitreal injections of ganciclovir (GCV). Case 2 was a 63-year-old woman who had a history of recurrent anterior uveitis in her left eye. Two years after cataract surgery, AC inflammation, diffuse vitreous haze, and periphlebitis had developed. CMV DNA was detected in the AC. Intravitreal injections of GCV and oral valganciclovir were administered, and ocular inflammation finally improved. Conclusions: and importance: We experienced two cases of CMV panuveitis in immunocompetent adults, both of which responded well to anti-viral therapies. Keywords: Cytomegalovirus, Panuveitis, Immunocompetent, Intravitreal injection, PCR

Purpose: To report the unique clinical findings of a case of Herpes Simplex Virus Type 2 herpetic retinitis manifesting as a large elevated subretinal lesion. Observations: A 26-year-old Hispanic male with no significant past medical history presented with a one-week history of right eye pain and endorsement of worsening vision. Ophthalmic examination of the right eye identified a markedly elevated white subretinal lesion with associated findings of vitritis and hypotony. Ultrasound biomicroscopy demonstrated a diffusely thickened choroid and confirmed the observed subretinal mass. Examination of the fellow left eye was largely unremarkable with the exception of lesions suggestive of inactive chorioretinal scars. Diagnostic vitrectomy and vitreous PCR (polymerase chain reaction) was positive only for HSV-2 (herpes simplex virus type 2) and verified by two independent laboratories. The observed subretinal lesion of right eye improved on intravenous acyclovir and intravitreal foscarnet treatment. Conclusions and Importance: Presented here is an unusual, novel clinical presentation of HSV-2 acute retinal necrosis manifesting as an elevated subretinal lesion along with findings of panuveitis. This case suggests that consideration should be given to the diagnosis of HSV ARN (acute retinal necrosis) when a subretinal elevation is concomitantly appreciated in the setting of vitritis and chorioretinal lesions. Keywords: Acute retinal necrosis, Herpes simplex virus type 2, Panuveitis, Polymerase chain reaction

Purpose To report the case of an immunocompetent adult presenting with endogenous Fusarium endophthalmitis. Observations A woman in her thirties presented with symptoms and signs of a unilateral anterior uveitis. After initial improvement with topical corticosteroids, she continued to develop a panuveitis with an associated drop in vision to counting fingers. A vitreous biopsy confirmed Fusarium solani by 18S rRNA fungal gene detection and PCR sequencing. Despite treatment with pars plana vitrectomy, intravitreal amphotericin B and systemic voriconazole her visual outcome was poor. Detailed review of her antecedent history revealed the route of acquisition to be a thorn prick to the hand two weeks prior to presentation. Conclusions and importance This patient's endophthalmitis most likely resulted from cutaneous inoculation of Fusarium solani with subsequent hematogenous spread. Endogenous Fusarium endophthalmitis is well recognized in the immunocompromised but is very rarely seen in the immunocompetent. This case highlights the importance of thorough history-taking and consideration of fungal endophthalmitis in the differential diagnosis of a treatment-refractory uveitis.

Purpose To report a rare case of Vogt-Koyanagi-Harada disease likely secondary to post-infectious Mycoplasma pneumoniae autoimmune response in a 14-year-old Hispanic female. Observations On presentation, visual acuity was 20/400 in the right eye and 20/20 in the left eye. The patient also had bilateral hyperemia, subretinal fluid, and vitreous cell graded at 1+. Fluorescein angiography and indocyanine green chorioangiography showed bilateral peripapillary hypofluorescence consistent with blocking and hyperflourescence consistent with staining. Laboratory testing showed elevated M. pneumoniae IgM and rising IgG antibodies. Topical steroids and oral steroids helped mitigate the systemic disease process and fully restore visual acuity through the 7-week mark. Conclusions and Importance The patient had elevated M. pneumoniae IgM and rising IgG antibodies resulting in ocular inflammation likely secondary to an autoimmune response. In this case of post-infectious M. pneumoniae, topical corticosteroids were beneficial in mitigating ocular manifestations initially, although oral steroids were needed and tapered over 6 weeks.

Purpose To report the ophthalmological approach of a patient with Blau syndrome (BS) in Colombia. Observations We describe a 9-year-old Colombian boy with sporadic BS due to a de novo nucleotide-binding oligomerization domain containing 2 (NOD2) mutation, who presented with joint and dermatologic symptoms. He was referred to the uveitis service with a single functional eye, due to retinal detachment in the other eye. Despite treatment with corticosteroids, methotrexate, and adalimumab, the patient continued to exhibit progressive disease. Conclusion BS-related uveitis is characterized by severe ocular morbidity. Appropriate interdisciplinary treatment is necessary for the correct identification and management of the disease, considering the inherent difficulty in its diagnosis due to its diverse clinical manifestations. The severity of BS-related uveitis in this report highlights the need for more effective therapies.

Purpose To report a case of bilateral uveitis secondary to intravenous nivolumab therapy in a patient with stage IV non-small cell lung cancer. Observations A 53-year-old male with stage IV non-small cell lung cancer presented with gradual onset of blurry vision in the left eye for nine days after completion of the first cycle of intravenous nivolumab chemotherapy. At initial presentation, best-corrected visual acuity was 20/25 in the right eye and 20/30 in the left eye. Slit lamp biomicroscopy examination of the left eye showed temporal injection of the conjunctiva and sclera, granulomatous keratic precipitates, and vitreous cells in the posterior segment. Imaging studies, including fundus photography, fluorescein angiography, fundus autofluorescence, optical coherence tomography, iridocyanine green angiography, and B scan ultrasonography, demonstrated acute inflammation in the posterior segment of the right eye and anterior, intermediate and posterior segments of the left eye. Nivolumab was discontinued and the patient received a course of corticosteroids resulting in resolution of visual complaints. The patient subsequently developed elevated and sustained intraocular pressures and decreased visual acuity in the left eye secondary to treatment complications. The patient was then lost to follow-up. Conclusions and Importance To our best knowledge, this is a rare case of bilateral uveitis secondary to intravenous nivolumab use and the sixteenth reported case of nivolumab-induced uveitis. Physicians should be aware of possible ocular complications associated with the use of nivolumab and provide prompt treatment when necessary.

Purpose: To report a case of a 28-years-old male who presented with a worsening unilateral panuveitis after intensive topical steroid therapy which resulted in a diagnostic and treatment dilemma as to whether the patient should be treated as for infective endophthalmitis despite no immediate known infective risk factors. Observations: A patient presented initially with unilateral non-granulomatous acute anterior uveitis which worsened after being started on intensive steroid therapy, developing fibrinous panuveitis. The rapid worsening of inflammation and vision deterioration despite being on intensive steroid therapy resulted in the patient subsequently being treated as for infective endophthalmitis. Anterior chamber and vitreous tap were done and intravitreal antibiotics were administered, along with topical antibiotics therapy. Vitrectomy was withheld due to the lack of conclusive evidence of infective etiology and risk factors. Full uveitis and infective workup were done. Investigations were largely unremarkable, and fluid and vitreous cultures were negative. HLA B27 was positive. The patient was subsequently started on systemic oral corticosteroids and improved in terms of his visual acuity, signs and symptoms. Conclusions: HLA B27-associated uveitis can present in a range of clinical spectrum with the extreme being of unusual severity in the form of fibrinous panuveitis that can mimic infective endophthalmitis. This can lead to a dilemma in management and subject patients to unnecessary risks from diagnostic and therapeutic interventions. Patients under this subgroup may require long term systemic immunosuppression therapy for disease remission and will require long term follow up. Keywords: HLA B27, Panuveitis, Infective endophthalmitis, Tap and inject

Purpose This is a retrospective case report illustrating the diagnostic and therapeutic challenges associated with a chronic rhegmatogenous retinal detachment masquerading as a severe panuveitis with intense anterior chamber inflammation. We have included clinical features, anterior segment and fundus photography, B-scan ultrasonography, fluorescein angiography, and intraoperative findings. Observations A 26-year-old male presented with features of unilateral panuveitis: hypotony, anterior segment inflammation (posterior synechiae and anterior chamber cell with fibrin clumping), diffuse choroidal thickening, and retinal detachment. Laboratory investigations for infectious or rheumatologic processes were negative, and empiric systemic corticosteroid therapy was unsuccessful. This prompted suspicion for an alternate primary etiology, and pars plana vitrectomy revealed small retinal breaks as the underlying cause of the retinal detachment and inflammation. Conclusions Rhegmatogenous retinal detachments are a known cause of intraocular inflammation. Nevertheless, it remains a challenge to recognize retinal breaks in this setting, particularly with robust anterior segment inflammation and posterior findings resembling severe exudative uveitis. Being aware of this unique presentation may prevent delays in diagnosis and have important prognostic implications.

Purpose: Seasonal Hyperacute Panuveitis (SHAPU) is a mysterious but rapidly devastating intraocular inflammatory disease reported only from Nepal during odd years after monsoon. Though it predominantly affects children, it has been reported between 9 months and 50 years age. But herein, we report a case of SHAPU in a 38-days-baby during 2017 SHAPU outbreak. Observations: Parents of a 38-days-healthy male baby from Pokhara noticed redness in child's right eye since 5 days (October 2017). They noticed the fall of a white moth from the tubelight over the child's face 1-day prior to symptom onset. On examination, both eyes followed and fixated at light. The right eye was congested with dense anterior chamber reaction and white pupillary reflex obscuring the glow of the retina. Ultrasonography showed hyperechoic vitreous shadows with choroidal thickening. Left eye was normal. The child underwent vitreous tap with core vitrectomy + lensectomy with intravitreal antibiotics and steroid ↓GA. The blood investigations were normal and the vitreous sample yielded no organism growth. Viral PCR analysis was negative. The intraocular inflammation decreased and fundal glow was visible. Conclusions: AND IMPORTANCE: SHAPU can occur in a child as young as 38 days. Link with moth remains the strongest suspect. Keywords: Intravitreal injection, Leukocoria, Moth, SHAPU, Vitrectomy

Purpose To report a case of bilateral panuveitis with multifocal choroiditis associated with a systemic sarcoidosis unmasked by pembrolizumab. Case report A 68-years old woman with a history of metastatic melanoma treated by pembrolizumab consulted for bilateral blurred vision. The ophthalmologic examination revealed a bilateral anterior uveitis with cells in the anterior chamber and granulomatous keratic precipitates, vitreous cells and multifocal choroiditis confirmed by indocyanine green angiography. The systemic workup revealed pulmonary sarcoidosis confirmed by biopsy. Conclusions and importance Pembrolizumab is an immune checkpoint inhibitor therapy used in the treatment of metastatic melanoma. We report a pembrolizumab-associated sarcoidosis revealed by a panuveitis with multifocal choroiditis. Physicians should be aware of the potential inflammatory and autoimmune disease that may be induced by immunomodulatory therapies.