Com Gulnur, Awad Samah, Trenor Cameron C
*Department of Pediatric Pulmonary Medicine, University of Southern California, Los Angeles, CA †Department of Pediatrics, Jordan University of Science and Technology, Irbid, Jordan ‡Division of Hematology/Oncology, Vascular Anomalies Center, Boston Children's Hospital §Harvard Medical School, Boston, MA.
J Pediatr Hematol Oncol. 2017 May;39(4):e236-e239. doi: 10.1097/MPH.0000000000000800.
Cutaneovisceral angiomatosis with thrombocytopenia (CAT), also called multifocal lymphangioendotheliomatosis with thrombocytopenia (MLT), is a rare and newly described vascular malformation. Skin manifestations and thrombocytopenia are the hallmark of CAT/MLT, and visceral lesions are described. We report an infant with pulmonary hemorrhage, thrombocytopenia, and antiplatelet antibodies. There was no cutaneous involvement and the child was initially diagnosed with immune thrombocytopenia. Poor response to immune thrombocytopenia-directed therapy raised suspicion for an alternative diagnosis, and the ultimate diagnosis of CAT/MLT was made by lung tissue sampling. Unexpectedly, 2 years after resolution of pulmonary lesions and thrombocytopenia, the child developed typical cutaneous lesions.
伴血小板减少的皮肤内脏血管瘤病（CAT），也称为伴血小板减少的多灶性淋巴管内皮瘤病（MLT），是一种罕见且新描述的血管畸形。皮肤表现和血小板减少是CAT/MLT的标志，且有内脏病变的描述。我们报告一名患有肺出血、血小板减少和抗血小板抗体的婴儿。无皮肤受累，该患儿最初被诊断为免疫性血小板减少症。对免疫性血小板减少症针对性治疗反应不佳引发了对另一种诊断的怀疑，最终通过肺组织取样确诊为CAT/MLT。出乎意料的是，肺部病变和血小板减少症消退2年后，该患儿出现了典型的皮肤病变。
