Not Just Sjögren's: A Rare Life-Threatening Manifestation in a Pediatric Patient

Macrophage activation syndrome (MAS), a severe hyperinflammatory state within the spectrum of hemophagocytic lymphohistiocytosis (HLH), is a life-threatening complication most commonly associated with rheumatologic disorders such as systemic juvenile idiopathic arthritis. Pediatric Sjögren's disease, however, is a rare and underrecognized etiology. We present the case of an eight-year-old female patient with no prior medical history who developed MAS in the setting of newly diagnosed Sjögren's disease, notably without classical sicca symptoms. Her course was marked by progressive systemic inflammation, multiorgan involvement, and laboratory findings suggestive of HLH/MAS. Diagnosis was confirmed through bone marrow biopsy and rheumatologic evaluation, and she responded favorably to immunosuppressive therapy with anakinra and corticosteroids. This case highlights the diagnostic challenges of MAS in atypical pediatric presentations and underscores the need for broader clinical suspicion in cytokine storm syndromes, particularly when established classification criteria are lacking.

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Publication Details

Journal
Cureus
Published
2025-11-21
DOI
https://doi.org/10.7759/cureus.97434
Primary Topic
Autoimmune and Inflammatory Disorders Research
Type
article
Field-Weighted Citation Impact
0.00
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article

Not Just Sjögren's: A Rare Life-Threatening Manifestation in a Pediatric Patient

Basil M. Fathalla, Tarek Husien, Abigail Cooley, Elise Dahan et al.
Cureus
Autoimmune and Inflammatory Disorders Research
article

Not Just Sjögren's: A Rare Life-Threatening Manifestation in a Pediatric Patient

Basil M. Fathalla, Tarek Husien, Abigail Cooley, Elise Dahan, Kendel Ridgeway
article en

Abstract

Macrophage activation syndrome (MAS), a severe hyperinflammatory state within the spectrum of hemophagocytic lymphohistiocytosis (HLH), is a life-threatening complication most commonly associated with rheumatologic disorders such as systemic juvenile idiopathic arthritis. Pediatric Sjögren's disease, however, is a rare and underrecognized etiology. We present the case of an eight-year-old female patient with no prior medical history who developed MAS in the setting of newly diagnosed Sjögren's disease, notably without classical sicca symptoms. Her course was marked by progressive systemic inflammation, multiorgan involvement, and laboratory findings suggestive of HLH/MAS. Diagnosis was confirmed through bone marrow biopsy and rheumatologic evaluation, and she responded favorably to immunosuppressive therapy with anakinra and corticosteroids. This case highlights the diagnostic challenges of MAS in atypical pediatric presentations and underscores the need for broader clinical suspicion in cytokine storm syndromes, particularly when established classification criteria are lacking.

CureusVol. 17(11)
Detroit Medical Center (US)
Openalex Percentile: Top 53%
Autoimmune and Inflammatory Disorders Research
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