Extensive choroidal infarction in a case of mixed essential cryoglobulinaemia in a postpartum female

Takkar, Brijesh; Azad, Shorya Vardhan; Kumar, Uma; Venkatesh, Pradeep

doi:10.1136/bcr-2016-216513

Cited by 5 publications

(6 citation statements)

References 22 publications

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“…Loss of capillary layers and dilated large vessel layer observed in the right eye of this case has been considered as a sign of previous acute choroidal injury [83]. The most plausible hypothesis explaining the development of ocular findings includes ischemia secondary to paraproteinemia-related hyperviscosity, cryoglobulin induced inflammation and occlusive process caused by the unusual temperature-dependent globulin solubility [83]. In IgA vasculitis (Henoch-Schönlein) characterized by abnormal IgA deposits affecting small vessels, rare cases of ocular involvement including central retinal artery or vein occlusion, anterior ischemic optic neuropathy, ischemic retinal vasculitis, scleritis and sclerokeratitis have been reported [84][85][86].…”

Section: Small Vessel Vasculitis (Svv)mentioning

confidence: 61%

“…An extensive triangular wedge-shaped choroidal infarction leading to irreversible visual loss has also been reported in a postpartum female with mixed essential cryoglobulinemia [83]. Loss of capillary layers and dilated large vessel layer observed in the right eye of this case has been considered as a sign of previous acute choroidal injury [83]. The most plausible hypothesis explaining the development of ocular findings includes ischemia secondary to paraproteinemia-related hyperviscosity, cryoglobulin induced inflammation and occlusive process caused by the unusual temperature-dependent globulin solubility [83].…”

Section: Small Vessel Vasculitis (Svv)mentioning

confidence: 79%

“…This may be attributed to the corticosteroid use in some of the cases or to the increased capillary permeability caused by excessive plasma protein concentration resulting with accumulation of subretinal and sub-RPE fluid [82]. An extensive triangular wedge-shaped choroidal infarction leading to irreversible visual loss has also been reported in a postpartum female with mixed essential cryoglobulinemia [83]. Loss of capillary layers and dilated large vessel layer observed in the right eye of this case has been considered as a sign of previous acute choroidal injury [83].…”

Section: Small Vessel Vasculitis (Svv)mentioning

confidence: 99%

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Choroidal involvement in systemic vasculitis: a systematic review

Özdal

Tuğal-Tutkun

2022

J Ophthal Inflamm Infect

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Systemic vasculitides are a large group of heterogeneous diseases characterized by inflammatory destruction of blood vessels targeting diverse organs and tissues including the eye. As the most vascularized layer of the eye, the choroid is expected to be affected in multiple systemic rheumatologic diseases with vascular involvement. While there are plenty of studies investigating retinal vascular involvement, choroidal vascular involvement in systemic vasculitides has not been investigated in isolation. However, choroidal manifestations including thickness changes, choroidal vasculitis and ischemia may be the earliest diagnostic features of systemic vasculitic diseases. Thus, multimodal imaging of the choroid may help early detection of choroidal involvement which may also have prognostic implications in these life-threatening diseases. This article aimed to review involvement of the choroid in systemic vasculitic diseases.

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Section: Small Vessel Vasculitis (Svv)mentioning

confidence: 61%

Section: Small Vessel Vasculitis (Svv)mentioning

confidence: 79%

Section: Small Vessel Vasculitis (Svv)mentioning

confidence: 99%

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Choroidal involvement in systemic vasculitis: a systematic review

Özdal

Tuğal-Tutkun

2022

J Ophthal Inflamm Infect

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“…Moreover, we exclude references with unconfirmed diagnosis [ 68 ], very uncertain timelines [ 69 ] or diagnosis made more than 6 months after delivery [ 70 ], because 6 months is considered as the maximal length of the post-partum period for some authors [ 71 ] and as the time delay during which the hormonal environment leading to ovulation has not been restored in more than 80% of breastfeeding women of Western countries [ 72 ]. The cases of vasculitis affecting only the neonate [ 73 ] were out of the scope of this review, as well as the cases of vasculitis related to an immunological process involving only the cerebral vasculature or the retina [ 74 , 75 ]. In these cases, other pathological processes such as vasospasm could not be ruled out as a differential diagnosis of vasculitis [ 76 – 78 ].…”

Section: Discussionmentioning

confidence: 99%

Systemic vasculitis diagnosed during the post-partum period: case report and review of the literature

Demotier

Orquevaux

N’Guyen

2023

matern health, neonatol and perinatol

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Introduction The vasculitis diagnosed specifically in the post-partum period are less well known. We report here such a case followed by a descriptive review of the literature. Case report A 25 year-old French nurse reported abrupt-onset musculoskeletal pain 15 days after delivery of her first infant. Her first pregnancy was uneventful. The physical examination yielded only bilateral conjunctivitis and purpuric eruption of lower limbs, and complementary investigations evidenced pulmonary renal syndrome in connection with the diagnosis of Granulomatosis with Polyangiitis. Methods We screened previous articles in Medline database using keywords (i) “post-partum” or “puerperium” (ii)”peripartum” (iii) “pregnancy” associated with “vasculitis”. Full texts were obtained from case reports or cases series whose title or abstract included keywords of interest (or synonyms). These references were secondarily excluded if the diagnosis of vasculitis was not confirmed or made before or more than 6 months after delivery and if vasculitis occurred only in the new born or affected only the cerebral vasculature or the retina. Results Fifty-six cases of vasculitis diagnosed in the post-partum period were included, 50 (89.3%) were secondary to an immunological process and 44 corresponded to primary vasculitis, 4 were secondary to Systemic Lupus Erythematosus, 1 to cryoglobulinaemia and 1 to cryoglobulinaemia associated with inflammatory bowel disease. The main primary vasculitis diagnosed were Takayasu Arteritis (n = 10), Eosinophilic granulomatosis with polyangiitis (n = 9), Granulomatosis with Polyangiitis (n = 7), Periarteritis Nodosa (n = 5) and Behcet’s disease (n = 4). The first symptom occurred before delivery in 26 (59.1%) and maternal death occurred in 4 (9.1%) out of the 44 primary vasculitis cases. Conclusion The vasculitis diagnosed in the post-partum period were mainly primary vasculitis among patients in whom the diagnosis had not been made during pregnancy. In less than half of cases and as described in ours, there were no reported symptoms before delivery. Therefore, the physicians should pay attention to symptoms and keep a high degree of suspicion for vasculitis before as well as after delivery.

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“…There have been no cohort studies aimed at establishing the prevalence and spectrum of ocular manifestations in patients with cryoglobulinemia. Several case reports have described the occurrence of the following ophthalmologic features in association with cryoglobulinemia: keratoconjunctivitis sicca [23,24]; PUK in patients with HCV- [25][26][27][28] or HBV- [29] related cryoglobulinemia; unilateral or bilateral central retinal vein occlusion [30,31]; massive choroidal infarction [32]; scleritis and retinal vasculitis [33]; anterior segment ischemia and neovascularization [34]; central serous chorioretinopathy [35,36]; Purtscher-like retinopathy [37][38][39]; mucosa-associated lymphoid tissue lymphoma of a lacrimal gland [40]; and funduscopic findings of HVS [41,42]. These reports well demonstrate that virtually all ocular tissues can be involved in CV.…”

Section: Discussionmentioning

confidence: 99%

Ocular Manifestations of Cryoglobulinemia: a Reappraisal

Dammacco¹,

Cimino²,

Simone³

et al. 2022

Preprint

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Background/Objectives: To describe frequency and type of ocular manifestations in patients with cryoglobulinemic vasculitis (CV), as well as management approaches and outcomes. Subjects/Methods: This was a retrospective, observational study of 57 patients who were diagnosed with CV at a single center and regularly underwent a comprehensive ocular assessment. Results: Ophthalmologic manifestations were recorded in 16 patients (28%). The diagnoses included dry eye disease and primary Sjögren syndrome in 5 and 2 patients, respectively; peripheral ulcerative keratitis and anterior scleritis in 1 patient each; hyperviscosity syndrome and hypertensive retinopathy in 2 patients each; and Purtscher-like retinopathy in 3 patients. Twelve patients (75%) were anti-HCV/HCV RNA-positive, 11 of whom achieved a sustained virologic response (SVR) following treatment with interferon-α2b plus ribavirin or direct-acting antivirals. All patients were treated with ocular lubricants. Systemic therapeutic measures, including glucocorticoids, immunosuppressive and biologic agents, induced the disappearance or ≥ 50% reduction of cryoglobulins and major signs of vasculitis in 11 patients (68.7%). In the remaining 5 patients (31.3%), cryoglobulins and CV manifestations remained unchanged or decreased by < 50%. The corresponding ophthalmologic assessment showed a variable degree of improvement in the ocular symptoms in all but 2 patients (87.5%). The best corrected visual acuity following treatment improved in 26 eyes, was unchanged in 3 eyes, and worsened in 3 eyes. Conclusions: Eye involvement is not a rare event in CV patients. A timely diagnosis and the correct employment of the available therapeutic measures may result in a favorable outcome of the ocular and extra-ocular manifestations.

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Extensive choroidal infarction in a case of mixed essential cryoglobulinaemia in a postpartum female

Cited by 5 publications

References 22 publications

Choroidal involvement in systemic vasculitis: a systematic review

Choroidal involvement in systemic vasculitis: a systematic review

Systemic vasculitis diagnosed during the post-partum period: case report and review of the literature

Ocular Manifestations of Cryoglobulinemia: a Reappraisal

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