Primary systemic vasculitis: treatment of
difficult cases
R Foster
2
, E Rosenthal
1
, S Marques
1
, P Vounotrypidis
1
, S Sangle
1
and D D’Cruz
1
*
1
Lupus Research Unit, The Rayne Institute, St Thomas’ Hospital, London, UK; and
2
Department of Immunology, St Thomas’ Hospital, London, UK
The primary systemic necrotizing vasculitides are a severe group of diseases, which untreated have a
high mortality. The majority respond to treatment with high dose steroids and cyclophosphamide,
however a significant proportion of those treated suffer morbidity due to the side effects of these
agents, and a number of patients are refractory to treatment. We review and discuss alternative and
emerging treatment options for patients who fail or cannot tolerate conventional therapy.
An interesting subgroup of patients with systemic vasculitis and antiphospholipid antibodies
provides an additional diagnostic and therapeutic challenge. We review what is known about this
subgroup, and suggest screening for antiphospholipid antibodies in all patients with systemic
vasculitis. Lupus (2006) 15, 143–147.
Key words: antiphospholipid antibodies; anti-TNF alpha therapy; mycophenolate mofetil;
rituximab; vasculitis
Lupus (2006) 15, 143–147
www.lupus-journal.com
REVIEW
Introduction
The vasculitides are a heterogeneous group of
uncommon diseases, characterized by inflammatory
cell infiltration and necrosis of blood vessel walls.
The primary systemic necrotizing vasculitides are
a severe group of diseases, of which the
ANCA-associated small-vessel vasculitides, namely
Wegener’s Granulomatosis, Churg Strauss Syndrome
and Microscopic PolyAngiitis are the best defined
and most studied subgroup. Untreated, these diseases
have a two-year mortality of up to 90%.
1
Since the introduction of cyclophosphamide and
high-dose corticosteroids by Fauci et al.
2
in the 1970s,
prognosis has greatly improved and disease is con-
trolled in 80–90% of patients. For those patients who
respond to treatment, the morbidity associated with
long-term steroids and cyclophosphamide ie, diabetes,
infection, bladder cancer, haemorrhagic cystitis, bone
marrow toxicity and infertility, dictates that exposure
to these drugs should be kept to a minimum and other
less toxic treatments sought. In those who do not
respond, an alternative, effective treatment is urgently
needed.
A subgroup of patients with systemic vasculitis who
are also positive for antiphospholipid antibodies,
are being increasingly reported in the literature.
These patients present an additional diagnostic and
therapeutic challenge in the treatment of systemic
vasculitides.
Conventional treatment of ANCA-associated
vasculitis (AAV)
Induction of remission
Conventional treatment of AAV comprises both an
induction and a maintenance phase. Standard induc-
tion therapy consists of high dose corticosteroids and
cyclophosphamide (CYC).
3
CYC can be given as a
daily oral or as a pulsed IV regime. Pulsed therapy
seems to be as effective at inducing remission as oral
therapy and is less toxic, but there is a slightly higher
risk of disease relapse.
4
Whether pulsed or oral, CYC therapy is associated
with significant side effects. An alternative to CYC
during the induction phase is methotrexate (MTX),
this has been shown to be as effective at inducing
remission as oral CYC in early, less severe vasculitis
© 2006 Edward Arnold (Publishers) Ltd 10.1191/0961203306lu2277rr
*Correspondence: David D’Cruz, Lupus Research Unit, The Rayne
Institute, St Thomas’ Hospital, Lambeth Palace Road, London, SE1 9LN,
UK. E-mail: david.d’cruz@kcl.ac.uk