Ocular Involvement in the Histiocytoses: A Literature Review with an Illustrative Case Series

Kim, Jennifer; Steeples, Laura R; Jones, Nicholas P.

doi:10.1080/09273948.2021.1936566

Cited by 3 publications

(1 citation statement)

References 103 publications

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“…Literature summary of published case reports[5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23] …”

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Ophthalmic artery ischemia in hemophagocytic lymphohistiocytosis: A case report and review of current literature

Gupta,

Shah,

ElHousseini

et al. 2024

J Case Rep Images Opthalmol

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Introduction: This case report describes the ocular manifestations of hemophagocytic lymphohistiocytosis in a 35-year-old male with suspected Adult-onset Still’s Disease. Documented ocular presentations of hemophagocytic lymphohistiocytosis are variable, and the pathophysiology remains unclear. A complete review of the current literature suggests that posterior segment findings, including retinal and vitreous hemorrhage, are most common. Case Report: Here we describe the first published case of bilateral combined choroidal and retinal ischemia in a patient with ocular hemophagocytic lymphohistiocytosis. This is suggestive of complete ophthalmic artery occlusion driving proliferative retinopathy. Mechanistic hypotheses include anemia, thrombocytopenia, coagulopathy, and histiocytic infiltration. Conclusion: In this case, pre-retinal hemorrhage, retinal hemorrhage, and vitreous hemorrhage were treated with panretinal photocoagulation, pars plana vitrectomy, and bevacizumab at different stages in each eye. Unusually for ocular hemophagocytic lymphohistiocytosis, the patient developed a unilateral tractional retinal detachment, later thought to be due to early administration bevacizumab prior to vitrectomy in the left eye. Final visual acuity in this patient remains poor, and worse in the left eye. We therefore suggest early vitrectomy to maintain adequate views of the fundus before bevacizumab or panretinal photocoagulation in this patient cohort.

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“…Literature summary of published case reports[5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23] …”

unclassified

Ophthalmic artery ischemia in hemophagocytic lymphohistiocytosis: A case report and review of current literature

Gupta,

Shah,

ElHousseini

et al. 2024

J Case Rep Images Opthalmol

View full text Add to dashboard Cite

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Re: Banks et al.: Ophthalmologic involvement in adults with histiocytic disorders: clinical presentation and treatment outcomes (Ophthalmology. 2023;130:77-86)

2023

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Ocular findings in patients with histiocytosis and association with clinical and molecular features

Francis,

Reiner,

Canestraro

et al. 2024

Br J Ophthalmol

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Background/aimsOcular manifestations of histiocytosis and their genetic underpinnings are poorly characterised. This study characterises ocular sites of histiocytosis, notate genetic alterations and correlates to histiocytosis clinical features including subtype and sites of disease.MethodsProspective registry-based study of predominantly adult histiocytosis patients at a single-institution tertiary referral centre. 180 eyes of 90 patients (46 males, 44 females) with histiocytosis (Erdheim-Chester disease 34, Rosai-Dorfman 20, xanthogranuloma 7, mixed histiocytosis 13, Langerhans cell histiocytosis (LCH) 15, ALK-positive histiocytosis 1). Ocular findings were categorised by the structure involved. Histiocytosis subtype, sites of disease and genetic status were correlated to ocular findings.ResultsOcular disease was present in more than half the histiocytosis patient cohort and occurred with other disease sites. Ocular findings were statistically significantly different across histiocytic subtypes with LCH subtypes having the lowest proportion of ocular findings (7%) and all other subtypes having rates of ocular findings which were five times that of patients with LCH (p=0.0009). Of patients with ocular findings, 41% of patients reported ocular symptoms and were significantly more in the group with ocular disease present versus those patients without ocular involvement. The presence of ocular findings was not statistically different byBRAFV600E,MAP2K1orRASisoform mutational status.ConclusionsOcular disease is a common feature of histiocytosis with significant visual symptomatology and occurrence in tandem with multisystem sites. Ocular findings vary by histiocytic subtype. The mutational profile of the cohort reflects known mutations in this clinical population, with no specific driver mutation associated with ocular disease.

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Ocular Involvement in the Histiocytoses: A Literature Review with an Illustrative Case Series

Cited by 3 publications

References 103 publications

Ophthalmic artery ischemia in hemophagocytic lymphohistiocytosis: A case report and review of current literature

Ophthalmic artery ischemia in hemophagocytic lymphohistiocytosis: A case report and review of current literature

Re: Banks et al.: Ophthalmologic involvement in adults with histiocytic disorders: clinical presentation and treatment outcomes (Ophthalmology. 2023;130:77-86)

Ocular findings in patients with histiocytosis and association with clinical and molecular features

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