Cardiovascular

Déjà vu.

TL;DR

A case report demonstrates the diagnostic continuum between hypereosinophilic syndrome (HES) and eosinophilic granulomatosis with polyangiitis (EGPA), with a patient initially diagnosed with HES being reclassified as ANCA-negative EGPA five years later upon relapse with biopsy-proven eosinophilic vasculitis.

Key Findings

A patient initially diagnosed with HES was reclassified as ANCA-negative EGPA five years after initial presentation when relapse revealed biopsy-proven eosinophilic vasculitis.

  • Patient was a male in his mid-20s at initial presentation
  • Initial presentation included fever, purpuric rash, neuropathy, marked eosinophilia, hepatic dysfunction, and extensive multisystem thrombosis
  • Initial biopsy revealed eosinophilic infiltration without vasculitis, supporting HES diagnosis
  • Relapse five years later included recurrent thrombosis, purpura, eosinophilia, and biopsy-proven eosinophilic vasculitis, prompting reclassification as ANCA-negative EGPA

The patient achieved initial remission from HES with corticosteroids and anticoagulation.

  • Treatment consisted of corticosteroids and anticoagulation
  • Remission was achieved following this treatment regimen
  • The patient subsequently relapsed 5 years after achieving remission
  • Relapse was characterized by recurrent thrombosis, purpura, and eosinophilia

Treatment with corticosteroids, rituximab, and anticoagulation resulted in sustained remission over 36 months following reclassification as ANCA-negative EGPA.

  • Regimen included corticosteroids, rituximab, and anticoagulation
  • Sustained remission was maintained over 36 months of follow-up
  • This treatment was initiated after biopsy-proven eosinophilic vasculitis confirmed EGPA reclassification
  • The case illustrates that appropriate immunosuppressive therapy depends on accurate disease classification

EGPA and HES share overlapping clinical features that make early distinction difficult, particularly in ANCA-negative disease where eosinophil-mediated injury may precede vasculitis.

  • ANCA-negative EGPA is particularly challenging to distinguish from HES at initial presentation
  • Eosinophil-mediated organ injury can occur before vasculitis becomes histologically apparent
  • The case highlights 'the diagnostic continuum between HES and EGPA'
  • The authors emphasize the need for long-term follow-up with reassessment of diagnosis in such cases

The case underscores the importance of long-term surveillance and diagnostic reassessment in patients with eosinophilic disorders, as the underlying diagnosis may evolve over time.

  • The interval between initial HES diagnosis and reclassification as EGPA was 5 years
  • Early recognition of evolving vasculitis is described as 'essential to guide appropriate immunosuppressive therapy'
  • The authors advocate for reassessment of diagnosis during follow-up in patients with eosinophilic conditions
  • The title 'Déjà vu' references the recurrence of similar symptoms leading to a revised diagnosis

What This Means

This research describes the case of a young man in his mid-20s who was initially diagnosed with hypereosinophilic syndrome (HES), a condition where abnormally high levels of eosinophils (a type of white blood cell) cause damage to organs. He had serious symptoms including fever, skin rash, nerve damage, liver problems, and dangerous blood clots throughout his body. A tissue biopsy showed eosinophil infiltration but no blood vessel inflammation (vasculitis), which supported the HES diagnosis. He was treated with steroids and blood thinners and went into remission, but five years later his symptoms returned. This time, his biopsy showed eosinophilic vasculitis — inflammation of the blood vessels — leading doctors to reclassify his diagnosis as eosinophilic granulomatosis with polyangiitis (EGPA), a rarer and related autoimmune condition. He was then treated with steroids, the immune-suppressing drug rituximab, and anticoagulants, achieving sustained remission over the following 36 months. This research suggests that HES and EGPA exist on a diagnostic continuum — meaning they may not always be clearly distinct diseases, and a patient's diagnosis can change over time as the disease evolves. In particular, when patients do not have a certain antibody marker (ANCA-negative cases), it can be very difficult to distinguish between HES and EGPA early on, because the characteristic blood vessel inflammation of EGPA may not yet be present at the time of initial biopsy. The case highlights that what appears to be HES at first can later declare itself as EGPA when vasculitis becomes apparent. The practical implication is that patients diagnosed with HES — especially those with multisystem involvement — may need long-term follow-up and periodic reassessment of their diagnosis. This research suggests that if symptoms return or evolve, repeat biopsy and diagnostic re-evaluation are critical, since the correct diagnosis determines the most appropriate treatment. Recognizing the transition from eosinophil-mediated injury to true vasculitis early could allow clinicians to initiate more targeted immunosuppressive therapies sooner.

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Citation

Agrawal A, Patankar A, Samant R. (2026). Déjà vu.. BMJ case reports. https://doi.org/10.1136/bcr-2026-273616