Chronic cytomegalovirus necrotizing retinitis in a patient with scleroderma and mixed connective tissue disease

López-Herrero, F.; Sánchez-Vicente, J.L.; Espiñeira-Periñán, M.A.; Morenas-Iglesias, J. de las; Franco-Ruedas, C.; Rueda-Rueda, T.

doi:10.1016/j.oftale.2020.06.006

Cited by 3 publications

(1 citation statement)

References 8 publications

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“…It can occur in patients on immunosuppressive therapy. There has been a single case report of CMV retinitis in an MCTD patient [ 10 ]. In our patient, with this new infection, we had to withhold her azathioprine.…”

Section: Discussionmentioning

confidence: 99%

Uncovering the Unusual: A Case of Mixed Connective Tissue Disease With Rare Presentation, Atypical Complications, and Therapeutic Dilemmas

Khot¹,

Patil²,

Rathod³

et al. 2023

Cureus

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Mixed connective tissue disease (MCTD) is an overlap syndrome characterized by features of systemic lupus erythematosus, scleroderma, and polymyositis, along with the presence of the U1RNP antibody. A 46-yearold female patient presented with severe anemia, cough, and breathlessness, and was diagnosed with cold agglutinin disease, a type of autoimmune hemolytic anemia (AIHA). Autoimmune workup revealed MCTD by positive antinuclear and U1RNP antibodies. She had bilateral miliary mottling on X-ray and a tree-in-bud appearance on high-resolution computed tomography of the thorax, which were suggestive of pulmonary tuberculosis. Standard therapy with steroids was not advisable. She was subsequently started on antituberculosis treatment (anti-Koch's therapy), followed by steroid therapy and immunosuppressive therapy after three weeks. The patient responded well to treatment, but after two months, she developed cytomegalovirus (CMV) retinitis. Adult-onset CMV disease may occur as a result of primary infection, reinfection, or activation of a latent infection. Although not directly related, it can occur as an atypical association in the setting of immunosuppressive therapy. Morbidity and mortality are significantly increased in this population secondary to infectious potentiation: immunosuppression causes infections, and infections cause AIHA. The management of MCTD and secondary AIHA and immunosuppression poses a therapeutic challenge.

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Section: Discussionmentioning

confidence: 99%

Uncovering the Unusual: A Case of Mixed Connective Tissue Disease With Rare Presentation, Atypical Complications, and Therapeutic Dilemmas

Khot¹,

Patil²,

Rathod³

et al. 2023

Cureus

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Hydroxychloroquine/mycophenolate/prednisone

2020

Reactions Weekly

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Cytomegalovirus chronic retinal necrosis with ganciclovir resistance: a case report

Xia,

Kantipudi,

Striebich

et al. 2024

J Ophthal Inflamm Infect

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Background Cytomegalovirus (CMV) chronic retinal necrosis (CRN) is a rare viral retinal infection that occurs in mildly immunocompromised people. It shares some features with both acute retinal necrosis and CMV retinitis. It is typically treated with combination intravitreal and systemic ganciclovir. We discuss the management of a case of CMV CRN with ganciclovir resistance. Case presentation An 80-year-old female presented with one month of blurry vision in the left eye. She was being treated with abatacept, methotrexate, and prednisone for rheumatoid arthritis. Examination revealed anterior chamber and vitreous cell along with peripheral retinal whitening. Fluorescein angiogram showed diffuse retinal non-perfusion. Aqueous fluid PCR testing returned positive for CMV. The retinitis was initially controlled with oral and intravitreal ganciclovir, but then recurred and progressed despite these therapies. Ganciclovir resistance was suspected and the patient was switched to intravitreal foscarnet injections, along with oral letermovir and leflunomide, which lead to resolution of the retinitis. The patient has now continued with letermovir and leflunomide for approximately 2.5 years without reactivation of the retinitis or need for further intravitreal anti-viral injections and with adequate control of her rheumatoid arthritis. Conclusion The incidence of CMV CRN may increase in the future as the use of non-cytotoxic immunosuppressive therapies that result in relatively mild immunosuppression also increases. Treatment with ganciclovir is effective but frequently leads to resistance, as in our case. In this situation, combination therapy with letermovir and leflunomide, particularly in the setting of rheumatoid arthritis where leflunomide can also have an anti-inflammatory effect, can be considered.

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Chronic cytomegalovirus necrotizing retinitis in a patient with scleroderma and mixed connective tissue disease

Cited by 3 publications

References 8 publications

Uncovering the Unusual: A Case of Mixed Connective Tissue Disease With Rare Presentation, Atypical Complications, and Therapeutic Dilemmas

Uncovering the Unusual: A Case of Mixed Connective Tissue Disease With Rare Presentation, Atypical Complications, and Therapeutic Dilemmas

Hydroxychloroquine/mycophenolate/prednisone

Cytomegalovirus chronic retinal necrosis with ganciclovir resistance: a case report

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