Hydroxychloroquine treatment for primary Sjogren's syndrome: a two year double blind crossover trial.

Kruize, Aike A.; Hené, Ronald J.; Kallenberg, Cees G. M.; Bijsterveld, O. P. Van; Heide, Agnes van der; Kater, L.; Bijlsma, J. W. J.

doi:10.1136/ard.52.5.360

Cited by 197 publications

(94 citation statements)

References 12 publications

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“…Hydroxychloroquineis beneficial in decreasing myalgia, fatigue and laboratory findings including an elevated erythrocyte sedimentation rate and hypergammaglobulinemia ( 1 1 ). Similar findings were reported in an open-label, double-blind study (12). However,no significant changesin salivaryor tear flowrates wereobserved.…”

Section: Discussionsupporting

confidence: 76%

Efficacy of Low-dose Prednisolone Maintenance for Saliva Production and Serological Abnormalities in Patients with Primary Sjoegren's syndrome.

1999

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Section: Discussionsupporting

confidence: 76%

Efficacy of Low-dose Prednisolone Maintenance for Saliva Production and Serological Abnormalities in Patients with Primary Sjoegren's syndrome.

1999

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“…The drug is reported to improve salivary gland function and oral moisture in some studies [13][14][15] to decrease dry eyes, [16] and to have no effect on disease manifestations in others [17]. A recent case report describes the efficacy of HCQ in two patients with alopecia totalis [18].…”

Section: Discussionmentioning

confidence: 99%

Hydroxychloroquine Reverses Alopecia in Autoimmune Polyendocrinopathy- Candidiasis-Ectodermal Dystrophy (APECED): A Case Report

Seton¹

2013

J Clin Cell Immunol

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Background: Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) is a rare, autosomal recessive disease characterized by the onset of mucocutaneous candidiasis in childhood, followed by hypoparathyroidism, and in a subset of patients, alopecia and other endocrinopathies and autoimmune diseases. There is no known treatment to mitigate the course of the disease. Goal:To describe the outcomes in a woman with APECED who presented later in her life with Sjogren's disease and alopecia totalis, and was treated with hydroxychloroquine (HCQ). HCQ was in addition to treatment regimens for mucocutaneous candidiasis with esophageal stricture, hypoparathyroidism, insulin-dependent diabetes, hypothyroidism, B12 deficiency, and autoimmune hepatitis. Methods:Office visits, laboratory studies, including genetic characterization of her disease; and review of available medical records. ManuscriptAutoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) is a rare, autosomal recessive disease classically characterized by the onset of mucocutaneous candidiasis in childhood, followed by hypoparathyroidism, and in a subset of patients, adrenal insufficiency. Dental complications such as enamel hypoplasia are well recognized, and other autoimmune diseases frequently mark the lives of these patients including alopecia, pernicious anemia, malabsorption syndromes, autoimmune hepatitis and keratoconjunctivitis. What is striking about the autoimmunity in APECED is its predilection to cause sequential dysfunction of endocrine glands, such that an individual may suffer from insulin-dependent diabetes, hypothyroidism and hypogonadism in addition to the hypoparathyroidism and adrenal insufficiency that herald this disorder. What is also striking is the relentless progression of disease that may burden an individual life [1,2]. Particularly disheartening in this illness may be the occurrence of alopecia.This case study reports the reversal of alopecia in a woman with APECED using hydroxychloroquine (HCQ), and describes the impact HCQ has in diminishing some other features of this disease. Started on HCQ in late 2007, she had gradual re-growth of her hair, easing of the sicca symptoms in her mouth, and improvement in other parameters of APECED. This is the first report of HCQ being used in this disorder, and of its efficacy. Case ReportE.L. is a 58-year-old white female, born in Boston in 1955 to parents with no evidence of consanguinity, or clear Finnish or European ancestry. One of 5 children, E.L shares this genetic imprint with two other siblings, although their manifestations are remarkably different and neither sibling has alopecia. E.L. remembers the onset of oral candidiasis in primary or middle school. Dental abnormalities (presumed incongruities in the enamel) are mentioned later in her records, when it is reported that she presented with tetany age 14 years (1970), and was diagnosed with hypoparathyroidism at that time. Her older brother had been identified with hypocalcemia (calcium 5.2 mg/ dL)...

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“…[64] It has been shown, however, that administration of HCQ did not resolve sicca signs and symptoms or extraglandular manifestations in pSS patients. [65,66] In both trials in pSS, serious AEs rarely occurred. Gottenberg et al reported a similar frequency of serious AEs in the HCQ group and in the placebo group during the first 24 weeks.…”

Section: Hydroxychloroquinementioning

confidence: 99%

Safety of treatments for primary Sjögren’s syndrome

Nimwegen

Moerman

Smitt

et al. 2016

Expert Opinion on Drug Safety

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Introduction: Primary Sjögren's syndrome (pSS) is a disabling auto-immune disease, affecting exocrine glands and several organs. Areas covered:In this review we analyze the safety of therapies used in pSS. Symptomatic treatment is widely applied due to the good supportive effect and good safety profile. Systemic stimulation of tears and saliva can be successful in pSS. However, cumbersome adverse events can influence the tolerability of this therapy. Evidence for the effectiveness of synthetic DMARDs therapies in pSS is limited, while there is a risk of adverse events. Several studies on biologic DMARD treatment of pSS patients have shown promising efficacy and safety results. Expert opinion: The safety of symptomatic treatment of pSS is very good. However, systemic therapy is necessary to achieve long-term relieve and prevention of organ-damage. Synthetic DMARDs have not shown much efficacy in earlier studies, and their benefits do not weigh up to the possible harms, while biologic DMARDs show promising results regarding efficacy and cause mostly mild adverse events. Many questions remain unanswered regarding safety of DMARDs in pSS. There is a need for well designed studies, in which safety should be evaluated in a uniform manner to be able to compare the results between studies.ARTICLE HISTORY

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Hydroxychloroquine treatment for primary Sjogren's syndrome: a two year double blind crossover trial.

Cited by 197 publications

References 12 publications

Efficacy of Low-dose Prednisolone Maintenance for Saliva Production and Serological Abnormalities in Patients with Primary Sjoegren's syndrome.

Efficacy of Low-dose Prednisolone Maintenance for Saliva Production and Serological Abnormalities in Patients with Primary Sjoegren's syndrome.

Hydroxychloroquine Reverses Alopecia in Autoimmune Polyendocrinopathy- Candidiasis-Ectodermal Dystrophy (APECED): A Case Report

Safety of treatments for primary Sjögren’s syndrome

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