Mishra Kirtisudha, Singla Shilpy, Batra Vineeta Vijay, Basu Srikanta, Kumar Praveen
Paediatr Int Child Health. 2013 Dec 6:2046905513Y0000000065. doi: 10.1179/2046905513Y.0000000065.
Cold agglutinin syndrome (CAS) secondary to infection is rare, usually presenting with anaemia and minor skin changes. A 12-year-old girl with secondary CAS associated with extensive cutaneous necrosis is reported. She presented with fever and multiple necrotic lesions over both cheeks, the tip of nose, ear margins, hands and buttocks, along with pallor, hepatospenomegaly, acrocyanosis and gangrene of the fifth digit of the right hand. She had anaemia, unconjugated hyperbilirubinaemia and a positive direct antiglobulin test owing to cold agglutinins of the IgM type with anti-i specificity and titres of 1:512 at 4°C. Results of bone marrow examination were normal and cryoglobulins were negative. Cold antibodies released even during a brief, self-limited febrile illness can cause widespread cutaneous gangrene. We believe this is the first report in the paediatric age group.
感染继发的冷凝集素综合征(CAS)较为罕见,通常表现为贫血和轻微的皮肤改变。本文报道了一名12岁患有继发CAS并伴有广泛皮肤坏死的女孩。她出现发热,双侧脸颊、鼻尖、耳缘、手部和臀部有多处坏死性病变,同时伴有面色苍白、肝脾肿大、肢端发绀以及右手第五指坏疽。她存在贫血、非结合性高胆红素血症,直接抗球蛋白试验呈阳性,原因是IgM型冷凝集素具有抗-i特异性,在4°C时滴度为1:512。骨髓检查结果正常,冷球蛋白为阴性。即使在短暂的自限性发热疾病期间释放的冷抗体也可导致广泛的皮肤坏疽。我们认为这是儿科年龄组中的首例报告。