Frosted branch angiitis due to cytomegalovirus-associated unmasking immune reconstitution inflammatory syndrome: a case report and literature review

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Abstract

Background

Cytomegalovirus (CMV) retinitis is a common opportunistic infection in patients with acquired immunodeficiency syndrome. The common funduscopic manifestations are haemorrhagic necrotising variety and granular variety. Frosted branch angiitis (FBA), as a special form, when it occurred after antiretroviral therapy(ART), could possibly be associated with immune reconstitution. We report a case of FBA secondary to CMV infection-associated unmasking immune reconstitution inflammatory syndrome (IRIS).

Case presentation

A 27-year-old man with human immunodeficiency virus infection developed FBA after 35 days of ART. The left Aqueous humour (AqH) tested positive for CMV DNA, and the patient was diagnosed with CMV retinitis. The degree of intraocular inflammation was reflected by increased levels of interleukin (IL)-6 and IL-8 in AqH. After anti-CMV treatment and continuous ART for several months, his FBA and vision significantly improved. CMV DNA became undetectable in the left AqH, and the IL-6 and IL-8 levels in AqH decreased.

Conclusion

FBA could be a sign of CMV-associated unmasking IRIS. Anti-CMV treatment alone or combination with steroid treatment may be administered, depending on the changes in CMV DNA load and immunologic profile of AqH.

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Most cited references 18

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Frosted branch angiitis: a review.

Adam Walker, Andrea N. Jones, A Iguchi (2004)

The purpose of this study is to present the first report of a case of primary frosted branch angiitis from the UK and to review the characteristics of this rare disease. Primary frosted branch angiitis causes characteristic florid translucent retinal perivascular sheathing of both arterioles and venules in association with variable uveitis, retinal oedema and visual loss, normally with good recovery. A total of 57 cases have been reported in the world literature. Atypical, typically focal frosted branch angiitis may also occur secondary to other causes of intraocular inflammation, especially cytomegalovirus retinitis. Primary frosted branch angiitis has a characteristic presentation but a variable course, typically affecting children or young adults. The disease is likely to represent a common immune pathway in response to multiple infective agents. The optimal treatment is unclear.

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Opportunistic infections and immune reconstitution inflammatory syndrome in HIV-1-infected adults in the combined antiretroviral therapy era: a comprehensive review.

Christian Manzardo, Alberto C Guardo, Emilio Letang … (2015)

Despite the availability of effective combined antiretroviral treatment, many patients still present with advanced HIV infection, often accompanied by an AIDS-defining disease. A subgroup of patients starting antiretroviral treatment under these clinical conditions may experience paradoxical worsening of their disease as a result of an exaggerated immune response towards an active (but also subclinical) infectious agent, despite an appropriate virological and immunological response to the treatment. This clinical condition, known as immune reconstitution inflammatory syndrome, may cause significant morbidity and even mortality if it is not promptly recognized and treated. This review updates current knowledge about the incidence, diagnostic criteria, risk factors, clinical manifestations, and management of opportunistic infections and immune reconstitution inflammatory syndrome in the combined antiretroviral treatment era.

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Intraocular viral and immune pathogenesis of immune recovery uveitis in patients with healed cytomegalovirus retinitis.

R C N Cubel Garcia, F Torriani, James Freeman … (2006)

To investigate immune and viral contributions to the pathogenesis of immune recovery uveitis (IRU), which presents as vitritis, macular edema, or formation of epiretinal membranes, and develops in patients with acquired immunodeficiency syndrome (AIDS) who experienced cytomegalovirus (CMV) retinitis before antiretroviral treatment (ART) induced immune reconstitution. Aqueous and vitreous fluids from patients with IRU, active CMV retinitis, and control human immunodeficiency virus (HIV)-negative, noninflamed eyes were compared for presence of cytokines IL-6, IL12, interferon gamma using enzyme-linked immunosorbent assay techniques, and CMV DNA (by polymerase chain reaction). IRU eyes (11 patients, 18 samples) had the highest levels of IL-12 (median 48 pg/mL), moderate levels of IL-6 (median 146 pg/mL), and low but significant interferon gamma (median 15 pg/mL), compared to controls (P < 0.01). All uveitis eyes tested (9/9) were CMV DNA negative. In contrast, active CMV retinitis eyes were CMV DNA positive, had higher levels of IL-6 (median 349 pg/mL) (25 patients, 41 samples) than both control (P = 0.0001) and uveitis eyes (P = 0.048), similar levels of interferon gamma (median 27 pg/mL) to uveitis eyes, but less IL-12 (median 0 pg/mL) than uveitis eyes. Inflammatory IRU can be differentiated from active CMV retinitis by the presence of IL-12, less IL-6, and absence of detectable CMV replication.

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Author and article information

Contributors

Ying Wen:

ORCID: http://orcid.org/0000-0002-9775-6351

wenying666466@163.com

Journal

Journal ID (nlm-ta): BMC Infect Dis

Journal ID (iso-abbrev): BMC Infect Dis

Title: BMC Infectious Diseases

Publisher: BioMed Central (London )

ISSN (Electronic): 1471-2334

Publication date (Electronic): 26 June 2021

Publication date PMC-release: 26 June 2021

Publication date Collection: 2021

Volume: 21

Electronic Location Identifier: 613

Affiliations

[1 ]GRID grid.412636.4, Infectious Diseases Department, , The First Affiliated Hospital of China Medical University, ; No. 155, Nanjing North Street, Heping District, Shenyang, 110001 Liaoning Province China

[2 ]GRID grid.412636.4, Department of Ophthalmology, , The First Affiliated Hospital of China Medical University, ; Shenyang, Liaoning Province China

[3 ]GRID grid.508217.9, Department of Gastroenterology, , The sixth People’s Hospital of Shenyang, ; Shenyang, Liaoning Province China

Author information

Ying Wen http://orcid.org/0000-0002-9775-6351

Article

Publisher ID: 6311

DOI: 10.1186/s12879-021-06311-4

PMC ID: 8236202

PubMed ID: 34174839

SO-VID: e2f6f159-1ea7-4549-b17e-02f0a7a32e72

License:

Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

History

Date received : 31 December 2020

Date accepted : 10 June 2021

Funding

Funded by: FundRef http://dx.doi.org/10.13039/501100012172, Double First Class University Plan;

Award ID: 3110119068 to W.Y.

Award Recipient : Ying Wen

Custom metadata

ScienceOpen disciplines: Infectious disease & Microbiology

Keywords: frosted branch angiitis,anti-cytomegalovirus treatment,unmasking immune reconstitution inflammatory syndrome,case report

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ScienceOpen disciplines: Infectious disease & Microbiology

Keywords: frosted branch angiitis, anti-cytomegalovirus treatment, unmasking immune reconstitution inflammatory syndrome, case report

Frosted branch angiitis due to cytomegalovirus-associated unmasking immune reconstitution inflammatory syndrome: a case report and literature review

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Abstract

Background

Case presentation

Conclusion

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Most cited references 18

Frosted branch angiitis: a review.

Opportunistic infections and immune reconstitution inflammatory syndrome in HIV-1-infected adults in the combined antiretroviral therapy era: a comprehensive review.

Intraocular viral and immune pathogenesis of immune recovery uveitis in patients with healed cytomegalovirus retinitis.

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