Primary Sjögren Syndrome in a Child with a Neuromyelitis Optica Spectrum Disorder

Kornitzer, Jeffrey; Kimura, Yukiko; Janow, Ginger

doi:10.3899/jrheum.151207

Cited by 12 publications

(6 citation statements)

References 15 publications

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“…Biologic treatments such as RTX and etanercept or infliximab were prescribed in a minority of patients. Of the nine patients that were prescribed RTX, two patients had a diagnosis of MALT lymphoma [ 22 ], three patients had NMOSD [ 26 , 29 ] and four adolescent patients had SS presenting with severe psychiatric symptoms [ 20 ].…”

Section: Resultsmentioning

confidence: 99%

“…There is no mention of response to RTX in two cases of NMOSD [ 29 ], while good clinical outcome was reported in the third case [ 26 ] ( Table 3 ). Of the four patients who were given RTX for SS with psychiatric involvement, three patients experienced significant improvement in symptoms and were able to be weaned off antipsychotics [ 20 ].…”

Section: Resultsmentioning

confidence: 99%

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Treatment strategies for Sjögren’s syndrome with childhood onset: a systematic review of the literature

et al. 2021

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Objectives Sjögren's Syndrome (SS) with childhood onset is a rare autoimmune disease characterised by heterogeneous presentation. The lack of validated classification criteria makes it challenging to diagnose. Evidence-based guidelines for treatment of juvenile SS are not available due to the rarity of disease and the paucity of research in this patient population. This systematic review aims to summarise and appraise the current literature focused on pharmacological strategies for management of SS with childhood onset. Methods PubMed and MEDLINE/Scopus databases up to December 2020 have been screened for suitable reports highlighting pharmacological treatment of SS with childhood onset using the PRISMA 2009 reporting checklist. Animal studies have been excluded. Results 43 studies (34 case reports, 8 mini case series and one pilot study) were eligible for analysis. The studies retrieved included girls in 88% (120/137) of cases and had very low confidence level. Hydroxychloroquine (HCQ) was prescribed for parotid swelling, as well as in association with methotrexate (MTX) and non-steroidal anti-inflammatory drugs (NSAIDS) in patients with arthritis and arthralgia. Corticosteroids such as long courses of oral prednisone and IV methylprednisolone were commonly prescribed for children with severe disease presentations. Rituximab was mainly indicated for MALT lymphoma, and renal and nervous system complications. Other conventional DMARDs were prescribed in selected cases with extra-glandular manifestations. Conclusion Various therapies are used for the management of juvenile SS and are prescribed based on expert clinician’s opinion. There are currently no good quality studies that allow clinical recommendations for treatment in SS with childhood onset.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Treatment strategies for Sjögren’s syndrome with childhood onset: a systematic review of the literature

et al. 2021

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“…In their report, Kornitzer et al refer to cases previously published by our group as unconfirmed cases of NMOSD due to a lack of NMO-IgG results(1). Here, we provide additional information regarding these cases as well as to report two new cases to highlight the co-occurrence of childhood Sjögren syndrome (SS) and NMOSD.…”

Section: To the Editormentioning

confidence: 96%

Pediatric Neuromyelitis Optica Spectrum Disorder and Sjögren Syndrome: More Common Than Previously Thought?

Gmuca

Mehta

2017

J Rheumatol

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“…Kornitze [3] Tables 18 Case 1 2 3 4 References/Year Matsui [2] Kornitzer [3] Arabshahi [4] Bogdanovic [5] Age ( References/Year DeGuzman [13] Skalova [14] Yoshida [15] Berman [16] Age ( Case reports…”

Section: Discussionmentioning

confidence: 99%

Nephrological disorders and neurological involvement in pediatric primary Sjogren syndrome: a case report and review of literature

Zhao

Chen²,

Zhu

et al. 2020

Preprint

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Background: Sjögren syndrome(SS) is a rare disease in pediatrics, and little attention has been paid to the clinical feature in these patients. To date, there are few cases concern about neurological and nephrological disorders in childhood Sjögren syndrome. We describe a case of Sjögren syndrome in a 12-year-old girl who developed neurological disorders and interstitial nephritis and review the literature currently available on this topic. Case presentation: A 12-year-old girl was admitted to our hospital for arthritis and glucosuria. She was required to do labial gland and renal biopsy, because the positive for anti-nuclear antibody and anti-Sjögren syndrome B(anti-SSB) antibody. Then the biopsy was performed revealing the lymphocytic infiltrate in the small area and renal tubular interstitial damage,thus the diagnosis of Sjögren syndrome with tubular interstitial damage was made. Three months later, she presented again with headache, fever, nausea, vomiting and was recovered without drug therapy. Based on the patient's medical history, laboratory and imaging examination, and treatment, we speculate that the disorders of the nervous system were caused by the Sjögren syndrome. The girl has stable renal function and no residual nervous system damage in the next 1.5 years, but she underwent low dose prednisone therapy because of persistent renal glucosuria. Conclusion s: Nephrological disorders and neurological involvement are rare manifestations of Sjögren syndrome in children, and rarely presented as the initial symptoms. It should be suspected in children presenting with unexplained renal diseases, neurological abnormalities, or unexplained fever. Although there is no guidelines on the diagnosis and treatment of children Sjögren syndrome are currently available, early recognition and the appropriate treatment of renal damage and neurologic involvement would improve prognosis and prevent complications. Keywords : Neurologic manifestations, Kidney diseases, Sjogren’s syndrome, Children

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Primary Sjögren Syndrome in a Child with a Neuromyelitis Optica Spectrum Disorder

Cited by 12 publications

References 15 publications

Treatment strategies for Sjögren’s syndrome with childhood onset: a systematic review of the literature

Treatment strategies for Sjögren’s syndrome with childhood onset: a systematic review of the literature

Pediatric Neuromyelitis Optica Spectrum Disorder and Sjögren Syndrome: More Common Than Previously Thought?

Nephrological disorders and neurological involvement in pediatric primary Sjogren syndrome: a case report and review of literature

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