Cryoglobulinemia
1.Cryoglobulin#
- <37℃會沉澱(不論是plasma or serum; 若只在plasma降溫會沉澱叫cryofibrinogen)
- Immunoglobulins or (Immunoglobulins+complements)
- 有cryoglobulinemia很少有cryoglobulinemia syndrome
- Prevalence:clinically-significant cryoglobulinemia:1/10W
- 但HCV 40-65%, HIV 15-20%, (HCV+HIV) 65%, AIR 20%有cryoglobulin(代表一大堆人有cryoglobulin卻沒有S/S)
- Prevalence:clinically-significant cryoglobulinemia:1/10W
- 有cryoglobulinemia很少有cryoglobulinemia syndrome
2.Types#
- Type I(10-15%)
- Monoclonal Ab:多為IgG or IgM; 但也可以是IgA or light chain
- 非IC deposit-->no decreased complement but可hyperviscosity syndrome
- From
- M(B cells系列):(MM, WM, CLL...) >>> MGUS
- Monoclonal Ab:多為IgG or IgM; 但也可以是IgA or light chain
- Type II(mixed cryoglobulinemia)(50-60%)
- Polyclonal IgG and monoclonal IgM(against the IgG; =rheumatoid factor)
- IC deposit in BV-->vasculitis(small vessel leukocytoclastic vasculitis)-->C4↓↓, C3↓
- From
- I(主):HAV, HBV(MGN, MPGN, cryoglobulinemia, IgAN), HCV(MPGN, cryoglobulinemia, MGN, MCD, collapsing FSGS, TMA, TIN, fibrillary GN, immunotactoid GN(?)), EBV(MGN, cryoglobulinemia, FSGS), CMV(cryoglobulinemia, collapsing FSGS), HIV(MCD, collapsing FSGS, MPGN/cryoglobulinemia, lupus-like GN, IgAN), B19(collapsing FSGS, cryoglobulinemia), IE, streptococcus, fungus, malaria(MPGN, cryoglobulinemia, MGN...), Ameba
- I:SLE, Sjogren, RA, PAN
- M:B cell lymphoma, solid cancers
- HCV>>>AIR>idiopathic>LPD>other infections
- Polyclonal IgG and monoclonal IgM(against the IgG; =rheumatoid factor)
- Type III(也是mixed cryoglobulinemia)(25-30%)
- Polyclonal IgG and polyclonal IgM
- IC deposit in BV-->vasculitis(small vessel leukocytoclastic vasculitis) -->C4↓↓, C3↓
- From
- I:as above, 尤其HCV(仍多)
- I::as above
- M:lymphoproliferative malignancies
- HCV>>AIR>>idiopathic>other infections
- Polyclonal IgG and polyclonal IgM
- 之所以HCV佔一大部分, 部分也跟liver disease/cirrhosis之後清不掉Ab有關
3.S/S of type I cryoglobulinemia#
- 大多沒症狀, 若有症狀多以hyperviscosity syndrome/thrombosis表現, 如Raynaud, Gangrene/acrocyanosis, livedo reticularis, purpura
- Arthralgia, renal, PNS也可不過比mixed cryoglobulinemia少見
4.S/S of mixed cryoglobulinemia(有症狀者的比例)(多為constitutional, non-specific S/S; 感覺就是一些慢性發炎,血管炎症狀)(mean 60 y/o, 多好好壞壞)#
- Meltzer's triad(25-30%)
- Weakness, palpable purpura, arthralgia
- Weakness and fatigue(80%)
- Lower limbs palpable purpura(80%), ulcers(10%)
- Raynaud's phenomenon(35%)
- Arthralgia(70%)(多下肢PIP, MCP, ankles, knees)/Myalgia(common); but arthritis/myositis少見
- PNS(polyneuropathy為主)(60%)
- Hepatosplenomegaly==>thrombocytopenia; lymphadenopathy
- Renal disease(20-30%)(as below)
- Increased NHL(B cell)(<10%, 多type II, 多occur after 5-10 yrs after diagnosis)
- 因為↑BAFF
- Sicca syndrome(30%)
- Rare:CNS, lung(small airways disease and impairment DLCO)
5. Renal disease#
-
Average 2.5yr after disease onset(0-28 yrs)
-
GN(可isolated proteinuria~nephrotic syndrome or nephritis syndrome...; 可ESRD)(和一般MPGN類似?不過似乎是indolent, slow course, 病人大多死於infection, cardiac diseases...)
- Microscopic hematuria + non-nephrotic range proteinuria +/- renal insufficiency:40%
- Nephrotic syndrome +/- renal insufficiency:20%
- Acute renal failure:10%
- AGN:15%
- HTN:65%
- CKD without significant U/A abnormalities:13%(?!!)
-
Pathology
- >80%為MPGN; others:vasculitis with fibrinoid necrosis, intraglomerular hyaline thrombi(小心SLE也可), mesangial proliferative GN (這些可以一起出現)
-
More specific for cryoglobulin-induced disease if(below)
- Intraluminal thrombi composed of precipitated cryoglobulins(PAS(+))
Capillary microthrombi in mixed cryoglobulinemia

-
- >80%為MPGN; others:vasculitis with fibrinoid necrosis, intraglomerular hyaline thrombi(小心SLE也可), mesangial proliferative GN (這些可以一起出現)
-
Diffuse IgM deposition in the capillary loops
-
Subendothelial deposits on EM that often have a clear substructure with a characteristic "fingerprint" pattern of cryoprecipitates(as below)(25-40nm, 所以要D/D immunotactoid GN; cryoglobulin的microtubules大多比較短)
- Subepithelial deposits are uncommon since the cryoglobulins are too large to pass GBM
Fingerprint pattern in mixed cryoglobulinemia

Cryoglobulinemic GN

6.Lab#
- 主要:RA factor(type II?), cryoglobulin, HBV titers, anti-HCV, HCV RNA, HIV, C3, C4, other AIR titers, IFE
- 【必考臨床檢驗 SOP】Cryoglobulin 採檢規範(PMID 23838352):
- 採血針筒與試管須預熱至 37°C, 採血後全程 37°C 恆溫運送並於 37°C 凝固離心(否則在分離前就先冷析出→偽陰性)
- 取得血清後置 4°C 靜置至少 72 小時(至 7 天), 測其沉降比例即 Cryocrit
- 偽性檢驗異常: 室溫下冷析出的沉澱物會被血液分析儀誤判 → 偽性白血球增多與偽性血小板增多 (pseudoleukocytosis / pseudothrombocytosis)
- 小心有cryoglobulin的人很少一部分才有症狀[Ex:HCV(+)者50%有cryoglobulin(+), 但cryoglobulin(+)的HCV Pts只有1/3有S/S]
- 有cryoglobulinemia S/S者未必可以測到有cryoglobulin(部分因為技術問題)
- 【必考臨床檢驗 SOP】Cryoglobulin 採檢規範(PMID 23838352):
- Tissue biopsy:leukocytoclastic vasculitis; occasionally cryoglobulins in BV lumen(→ 見上段採檢規範偽性血球異常)
7.D/D#
- Other vasculitis/thromboembolism disease:HSP, ANCA, AIR, APS, TMA, Rickettsia, Malaria(本來就可以MPGN, cryoglobulinemia, MGN), Babesiosis, atrial myxoma...
8.Tx#
- Cold avoidance and analgesics
- Type I
- Tx of underlying
- Type II-III
- If idiopathic
- If mild to moderate manifestation
- Colchicine(1-2mg/day) or low dose steroid(10-25mg/day, taper to <10mg/day by 6M if feasible)
- if severe(any):severe digital ischemia/vasculitis, RPGN/nephrotic syndrome, GI vasculitis, pulmonary vasculitis, CNS vasculitis, severe neuropathy, heart failure
- Rituximab(alternative:Endoxan) + rapid tapered Steroid
- 註:Rituximab打贏Endoxan
- Add plasmapheresis (if life threatening or hyperviscosity syndrome)
- Rituximab(alternative:Endoxan) + rapid tapered Steroid
- If mild to moderate manifestation
- If underlying(+)
- If mild to moderate: 治療潛在病因即可(HCV 給予 DAA 抗病毒)
- If severe: 治療潛在病因 + Rituximab(alternative:Endoxan) + rapid tapered Steroid ± plasmapheresis(若危及生命或高黏滯度)(HBV 先以 Entecavir 覆蓋、HIV 啟動 ART)
- 【治療核心金句】HCV-cryo(KDIGO 2022; PMID 36411019): DAA 為治癒 HCV 冷凝球蛋白血症之第一線基石(典型臨床證據者啟動 DAA 前不再強制腎切片);若合併重症/器官威脅(RPGN、nephrotic、下肢壞死潰瘍),應立即聯合 Rituximab + 短療程類固醇(± 危及生命時血漿置換)與 DAA。現代 DAA 無免疫刺激風險,不需像過去干擾素時代延後投藥。
- 註:TPE/DFPP studies多用在MPGN, neuropathy, arthralgia, ulcerating purpura
- Uptodate建議:hyperviscosity syndrome, crescent GN, intestinal vasculitis, pulmonary hemorrhage, severe/refractory skin ulcers者併用TPE/DFPP
- If idiopathic