Multicentric Castleman’s disease (MCD), a polyclonal lymphoproliferative disorder of unknown aetiology, is a well-recognised complication of HIV disease. We present a case of MCD in an HIV-positive patient that is unusual on two counts: our patient’s MCD first presented in the context of an immune restoration inflammatory syndrome (IRIS), following the initiation of highly active antiretroviral therapy (HAART). In addition, her MCD was associated with the unusual complication of acquired angioedema (AAE), which resolved following treatment of the MCD. While AAE is frequently found to have an underlying diagnosis of a lymphoproliferative disease, this is the first reported case linking AAE to MCD.
MylonaEEBaraboutisIGLekakisLJ. Multicentric Castleman's disease in HIV infection: a systematic review of the literature. AIDS Rev2008; 10(1): 25–35.
2.
HenggeURRuzickaTTyringSK. Update on Kaposi’s sarcoma and other HHV8 associated diseases. Part 2: pathogenesis, Castleman’s disease, and pleural effusion lymphoma. Lancet Infect Dis2002; 2(6): 44–52.
3.
SayerRPaulJTukePW. Can plasma HHV8 viral load be used to differentiate multicentric Castleman disease from Kaposi sarcoma?Int J STD AIDS2011; 22(10): 585–589.
4.
ZietzCBognerJRGoebelF. An unusual cluster of cases of Castleman's disease during highly active antiretroviral therapy for AIDS. NEJM1999; 340: 1923–1924.
5.
LeeSMEdwardsSGChiltonDN. Highly active antiretroviral therapy alone may be an effective treatment for HIV-associated multicentric Castleman’s disease. Haematologica2010; 95: 1979–1981.
6.
MüllerMWandelSColebundersR. Immune reconstitution inflammatory syndrome in patients starting antiretroviral therapy for HIV infection: a systematic review and meta-analysis. Lancet Infect Dis2010; 10(4): 251–261.
StebbingJPantanowitzLDayyaniF. HIV-associated multicentric Castleman's disease. Am J Haematol2008; 83(6): 498–503.
9.
OksenhendlerEDuarteMSoulierJ. Multicentric Castleman’s disease in HIV infection: a clinical and pathological study of 20 patients. AIDS1996; 10: 61–67.
10.
CugnoMCastelliRCicardiM. Angioedema due to acquired C1-inhibitor deficiency: a bridging condition between autoimmunity and lymphoproliferation. Autoimmun Rev2008; 8(2): 156–159.
11.
FrigasE. Angioedema with acquired deficiency of the C1 inhibitor: a constellation of syndromes. Mayo Clin Proc1989; 64(10): 1269–1275.
