Successful full-term pregnancy in a woman with Cogan’s syndrome: a case report

Deliveliotou, Aikaterini; Moustakarias, Theodore; Argeitis, John; Vaggos, George; Vitoratos, N.; Hassiakos, Dimitrios

doi:10.1007/s10067-007-0664-4

Cited by 9 publications

(3 citation statements)

References 15 publications

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“…A search of the literature revealed only three other reports of pregnancy in combination with CS to date 12–14. Apart from one case of uncomplicated interstitial keratitis similar to the case reported here, all three pregnancies were uneventful.…”

Section: Discussionsupporting

confidence: 49%

Off-label application of intravenous immunoglobulin (IVIG) for treatment of Cogan’s syndrome during pregnancy

Scherg

Haag

Krieger³

2019

BMJ Case Rep

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We report the case of a woman with Cogan’s syndrome concomitant with the wish to have children. After three major flares of the disease that led to unilateral deafness, immunosuppressive therapy with prednisolone and azathioprine was started. Because of the severe side effects, an off-label therapy with intravenous immunoglobulin (IVIG) was initiated, under which our patient has since given birth to three healthy children. To our knowledge this is the first report to describe Cogan’s syndrome with multiple successful pregnancies under IVIG treatment.

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Section: Discussionsupporting

confidence: 49%

Off-label application of intravenous immunoglobulin (IVIG) for treatment of Cogan’s syndrome during pregnancy

Scherg

Haag

Krieger³

2019

BMJ Case Rep

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“…Furthermore, in this cohort, two out of the three miscarriage occurs in the same women who had Cogan syndrome, a rare and severe autoimmune disease. In the literature, only eight cases of successful pregnancy with this disease have been reported [ 86 , 87 , 88 , 89 , 90 , 91 ]. Thus, it cannot be concluded that the therapy caused pregnancy loss in these women.…”

Section: Discussionmentioning

confidence: 99%

A Systematic Review of the Safety of Blocking the IL-1 System in Human Pregnancy

Brien

Gaudreault

Hughes

et al. 2021

JCM

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Blockade of the interleukin-1 (IL-1) pathway has been used therapeutically in several inflammatory diseases including arthritis and cryopyrin-associated periodic syndrome (CAPS). These conditions frequently affect women of childbearing age and continued usage of IL-1 specific treatments throughout pregnancy has been reported. IL-1 is involved in pregnancy complications and its blockade could have therapeutic potential. We systematically reviewed all reported cases of IL-1 blockade in human pregnancy to assess safety and perinatal outcomes. We searched several databases to find reports of specific blockade of the IL-1 pathway at any stage of pregnancy, excluding broad spectrum or non-specific anti-inflammatory intervention. Our literature search generated 2439 references of which 22 studies included, following extensive review. From these, 88 different pregnancies were assessed. Most (64.8%) resulted in healthy term deliveries without any obstetrical/neonatal complications. Including pregnancy exposed to Anakinra or Canakinumab, 12 (15.0%) resulted in preterm birth and one stillbirth occurred. Regarding neonatal complications, 2 cases of renal agenesis (2.5%) were observed, and 6 infants were diagnosed with CAPS (7.5%). In conclusion, this systematic review describes that IL-1 blockade during pregnancy is not associated with increased adverse perinatal outcomes, considering that treated women all presented an inflammatory disease associated with elevated risk of pregnancy complications.

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“…Half of previously reported CS patients did not need any systemic immunosuppressive treatments throughout pregnancy, instead intermittent topical treatment with ophthalmic steroids was sufficient ( 10 – 13 ). Other pregnant patients in remission were successfully treated with hydroxychloroquine, or a combination therapy of either cyclosporine or intravenous immunoglobulin application (IVIG) with azathioprine and prednisolone ( 8 , 9 , 12 ).…”

Section: Discussionmentioning

confidence: 99%

Case Report: Effective and Safe Treatment With Certolizumab Pegol in Pregnant Patients With Cogan’s Syndrome: A Report of Three Pregnancies in Two Patients

et al. 2021

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Cogan’s syndrome is a rare autoimmune disease characterized by ocular inflammation and audiovestibular manifestations. Treatment consists of systemic glucocorticoids and other immunosuppressive agents including methotrexate, cyclophosphamide and TNF-α-inhibitors. Due to potential ovarian or fetal toxicity immunosuppressive treatment options are limited during pregnancies. Thus far there is a paucity of reports on pregnancies in Cogan’s syndrome. With minimal transplacental transfer, Certolizumab pegol is considered to be safe for the use in pregnant patients with underlying inflammatory diseases. However, there is no literature on the use of this TNF-α-inhibitor in Cogan’s syndrome in general and especially during gestation. Here we report three pregnancies in two Cogan’s Syndrome-patients treated with Certolizumab pegol. Treatment with Certolizumab pegol was effective and well tolerated in patients with Cogan’s syndrome and seems to be a safe treatment option during pregnancy.

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Successful full-term pregnancy in a woman with Cogan’s syndrome: a case report

Cited by 9 publications

References 15 publications

Off-label application of intravenous immunoglobulin (IVIG) for treatment of Cogan’s syndrome during pregnancy

Off-label application of intravenous immunoglobulin (IVIG) for treatment of Cogan’s syndrome during pregnancy

A Systematic Review of the Safety of Blocking the IL-1 System in Human Pregnancy

Case Report: Effective and Safe Treatment With Certolizumab Pegol in Pregnant Patients With Cogan’s Syndrome: A Report of Three Pregnancies in Two Patients

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