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Rheumatology Vol. II, Case ORCT-03 — Other Rheumatic and Connective Tissue Disorders

Systemic Sjögren's Disease: An Available Therapy Against a Better-Proven One

A single patient whose Sjögren's disease has moved from dryness into her kidneys and skin. The disagreement is between the drug that's available today and the drug with the cleaner trial behind it.

Abbreviations, terms, and other agents mentioned in this case SjD — Sjögren's disease  ·  ESSDAI — EULAR Sjögren's Syndrome Disease Activity Index  ·  ADCC — antibody-dependent cellular cytotoxicity  ·  BAFF-R — B-cell activating factor receptor  ·  CD40L — CD40 ligand
Presentation

Linda F., a 58-year-old woman who helps raise her two young grandchildren several afternoons a week while their parents work, was diagnosed with primary Sjögren's disease five years ago after years of dry eyes and dry mouth finally got a name. For most of that time it stayed a sicca problem, managed on artificial tears and pilocarpine, with her hypertension the more active line on her chart. Over the past six months that changed: recurrent low-grade fevers, new parotid gland swelling on both sides, and palpable purpura across both lower legs that a skin biopsy confirmed as small-vessel vasculitis. A kidney biopsy prompted by a mildly rising creatinine found mild tubulointerstitial nephritis — real, new organ involvement, not just an uncomfortable dry mouth.

The therapeutic literature on this exact moment is more conflicted than it looks from a distance. TEARS and TRACTISS, the two largest randomized trials of rituximab in Sjögren's, both missed their primary endpoints — but both were run in broader populations not specifically enriched for significant extraglandular disease, weighted more toward dryness and fatigue than toward the renal and vasculitic activity she now has. Whether that population mismatch means rituximab's real effect was diluted rather than absent, or whether it simply doesn't work as well as hoped, is exactly the question a reasonable specialist could read either way. Meanwhile, ianalumab's twin phase 3 trials, NEPTUNUS-1 and NEPTUNUS-2, reported the first positive, replicate primary-endpoint results for any systemic Sjögren's therapy — built specifically around ESSDAI at week 48, a systemic disease-activity index, which is the exact axis on which she is now active. The effect sizes are real but modest: ESSDAI fell 6.4 points against 5.1 on placebo in NEPTUNUS-1, and 6.5 against 5.5 in NEPTUNUS-2, and NEPTUNUS-2's patient-reported outcomes did not reach significance where NEPTUNUS-1's did. Ianalumab holds FDA Breakthrough Therapy designation granted in January 2026, with submission underway but no approval and no commercial availability; dazodalibep's phase 2 data are positive too, further behind in development. The asymmetry that decides today is therefore not which drug is better — it is that one of them can be started this afternoon and the other cannot, and her kidney is accruing injury on whichever timetable the argument takes.

Linda F. · 58 New Extraglandular Flare
History
Primary SjD 5 years, previously sicca-predominant; hypertension
New findings
Bilateral parotid swelling, palpable purpura (biopsy-confirmed small-vessel vasculitis)
Renal
Creatinine mildly elevated; biopsy shows mild tubulointerstitial nephritis
Therapy so far
Artificial tears, pilocarpine — sicca-directed only
Constitutional
Recurrent low-grade fevers over 6 months
Prior systemic therapy
None — first extraglandular escalation

In clinic, new renal and cutaneous disease on a five-year sicca history

Rheumatologist (Community Practice) Opening

TEARS and TRACTISS are the two trials everyone cites against rituximab in Sjögren's, and both missed their primary endpoints — but neither trial was enriched for patients with significant extraglandular disease. Her renal and cutaneous vasculitic findings put her in a phenotype where real-world use and subgroup data support rituximab despite those negative headline results. I want to start it now.

Rheumatologist (Clinical Trials) Response

You're right that neither trial was enriched for systemic disease, and that's a fair critique of how much weight their negative results should carry for a patient like her.

But NEPTUNUS-1 and NEPTUNUS-2 are worth naming directly: two large, replicate, positive phase 3 trials with a primary endpoint built on ESSDAI at week 48 — a systemic activity measure that maps far more directly onto her actual presentation than either older trial's dryness-and-fatigue-weighted composite. I'll concede the size of it: 6.4 points against 5.1, and 6.5 against 5.5. That is a modest separation, and NEPTUNUS-2's patient-reported outcomes missed significance entirely. "Available today" isn't the same claim as "proven to work," and choosing the weaker-evidence option because it's easier to get isn't obviously the safer call for a patient with active kidney disease.

Nephrologist Final

I want to step outside the biologic argument for a moment. Whichever of you is right, her interstitial nephritis needs glucocorticoids starting today, with mycophenolate close behind if the biopsy activity index supports it.

Whether the pathway forward is rituximab now or pursuing ianalumab through an early-access or trial route, the renal-directed therapy shouldn't wait on that decision being settled. A delay chasing the more evidence-based biologic would be a real, avoidable cost to her kidneys specifically — that part of this isn't actually in dispute between the two of you.

Regimen selected
Rituximab
Anti-CD20 Monoclonal Antibody · IV, systemic track
Started given current accessibility and her extraglandular phenotype, despite TEARS and TRACTISS's negative primary endpoints in broader, less systemically-active populations.
Prednisone (renal induction)
Glucocorticoid · Oral, started immediately
Started regardless of the biologic decision, given active biopsy-confirmed tubulointerstitial nephritis that cannot wait on that argument being settled.
Mycophenolate Mofetil
Immunosuppressant · Contingent on renal biopsy activity
Held ready pending the biopsy's full activity index, to be added alongside glucocorticoids if renal activity supports it.
Ianalumab — Considered, Not Yet Accessible
BAFF-R-Directed Monoclonal · Phase 3 positive, not FDA-approved
Positive replicate phase 3 data on systemic disease activity; Breakthrough Therapy designation granted, submission pending — an early-access or trial pathway to be explored in parallel.
Where this was left

Agreed: start glucocorticoids for renal induction today regardless of the biologic decision, begin rituximab in parallel given its accessibility and her active phenotype, and pursue discussion of an ianalumab trial or early-access pathway as a next step should the rituximab response prove inadequate.

Not agreed: whether ianalumab's cleaner trial data should have been pursued as the first-line systemic agent if access allowed, or whether rituximab's real-world track record in exactly this phenotype makes that question largely academic. The community and clinical-trials rheumatologists genuinely differ here, and neither treats the other's position as wrong.

Educational content only — a composite teaching case, not a real patient encounter or a substitute for clinical guidance. About These Cases →