{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,13]],"date-time":"2026-06-13T04:55:54Z","timestamp":1781326554360,"version":"3.54.1"},"reference-count":38,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,6,12]],"date-time":"2026-06-12T00:00:00Z","timestamp":1781222400000},"content-version":"vor","delay-in-days":162,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Case Reports in Hematology"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:p>Kaposiform hemangioendothelioma (KHE) is a rare vascular neoplasm that typically presents in infancy or early childhood, classified as a locally aggressive\/borderline vascular tumor. It most commonly involves superficial and deep soft tissues, often demonstrating infiltrative growth into adjacent muscle and bone, whereas retroperitoneal or intraabdominal presentations are less frequent. KHE is frequently complicated by Kasabach\u2013Merritt phenomenon (KMP), a severe consumptive coagulopathy characterized by profound thrombocytopenia, microangiopathic hemolytic anemia, and hypofibrinogenemia with life\u2010threatening sequelae. We report a case of a male neonate who developed KMP secondary to an abdominal KHE without overlying cutaneous involvement. Diagnosis was established through integration of clinical features, imaging findings, hematological evaluation, and confirmatory biopsy. This patient was managed with a multimodal regimen of sirolimus, corticosteroids, and vincristine, resulting in significant clinical improvement. This case underscores both the diagnostic complexity and therapeutic challenges of KHE complicated by KMP, particularly when lesions occur in deep anatomical sites. Given the rarity of this condition, current management strategies rely largely on expert consensus and case\u2010based experience, though recent randomized trial and consensus have established sirolimus\u2010based regimens as highly effective first\u2010line therapy.<\/jats:p>","DOI":"10.1155\/crh\/5825111","type":"journal-article","created":{"date-parts":[[2026,6,13]],"date-time":"2026-06-13T04:06:06Z","timestamp":1781323566000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["A Rare Case of Neonatal Kaposiform Hemangioendothelioma With Kasabach\u2013Merritt Phenomenon Presenting as an Abdominal Mass Without Cutaneous Manifestations"],"prefix":"10.1155","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0009-0001-5995-9123","authenticated-orcid":false,"given":"Su Young","family":"Park","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-6791-121X","authenticated-orcid":false,"given":"Amrryn","family":"Halari","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-2520-5988","authenticated-orcid":false,"given":"Jessenia","family":"Palacio-Meadows","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4746-1168","authenticated-orcid":false,"given":"Nehal S.","family":"Parikh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0003-2212-1487","authenticated-orcid":false,"given":"Danielle","family":"Bentsen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7467-5874","authenticated-orcid":false,"given":"Surasak","family":"Puvabanditsin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,6,12]]},"reference":[{"key":"e_1_2_11_1_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2012.06.044"},{"key":"e_1_2_11_2_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00432-018-2759-5"},{"key":"e_1_2_11_3_2","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2022-056293"},{"key":"e_1_2_11_4_2","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2024.1336763"},{"key":"e_1_2_11_5_2","doi-asserted-by":"publisher","DOI":"10.1186\/s12887-021-02573-8"},{"key":"e_1_2_11_6_2","doi-asserted-by":"publisher","DOI":"10.12788\/j.sder.2016.048"},{"key":"e_1_2_11_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.heliyon.2023.e15425"},{"key":"e_1_2_11_8_2","doi-asserted-by":"publisher","DOI":"10.1111\/bjd.16601"},{"key":"e_1_2_11_9_2","doi-asserted-by":"publisher","DOI":"10.1016\/s0022-3476(97)70249-x"},{"key":"e_1_2_11_10_2","doi-asserted-by":"publisher","DOI":"10.1097\/00006534-199711000-00001"},{"key":"e_1_2_11_11_2","doi-asserted-by":"publisher","DOI":"10.1002\/1545-5017.70086"},{"key":"e_1_2_11_12_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejim.2008.06.008"},{"key":"e_1_2_11_13_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.30779"},{"key":"e_1_2_11_14_2","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.35344"},{"key":"e_1_2_11_15_2","doi-asserted-by":"publisher","DOI":"10.1259\/bjr.20170798"},{"key":"e_1_2_11_16_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00431-025-06631-6"},{"key":"e_1_2_11_17_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.pathol.2017.03.001"},{"key":"e_1_2_11_18_2","doi-asserted-by":"publisher","DOI":"10.1111\/pde.15262"},{"key":"e_1_2_11_19_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2021014027"},{"key":"e_1_2_11_20_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-020-1320-1"},{"key":"e_1_2_11_21_2","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-199304000-00001"},{"key":"e_1_2_11_22_2","doi-asserted-by":"publisher","DOI":"10.1097\/00000478-200405000-00001"},{"key":"e_1_2_11_23_2","doi-asserted-by":"publisher","DOI":"10.1186\/1477-7819-9-57"},{"key":"e_1_2_11_24_2","doi-asserted-by":"publisher","DOI":"10.1309\/96U1-6LRR-AN5H-WWVE"},{"key":"e_1_2_11_25_2","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.3800444"},{"key":"e_1_2_11_26_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199907153410307"},{"key":"e_1_2_11_27_2","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.13557"},{"key":"e_1_2_11_28_2","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.30216"},{"key":"e_1_2_11_29_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.25296"},{"key":"e_1_2_11_30_2","unstructured":"National Comprehensive Cancer Network NCCN Clinical Practice Guidelines in Oncology (NCCN Guidelines\u00ae): Pediatric Acute Lymphoblastic Leukemia. Version 1.2025 Plymouth Meeting PA 2025 National Comprehensive Cancer Network."},{"key":"e_1_2_11_31_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIR.0000000000000641"},{"key":"e_1_2_11_32_2","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(19841101)54:9<2006::aid-cncr2820540937>3.0.co;2-f"},{"key":"e_1_2_11_33_2","doi-asserted-by":"publisher","DOI":"10.1007\/BF00273423"},{"key":"e_1_2_11_34_2","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2004.08.075"},{"key":"e_1_2_11_35_2","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.35207"},{"key":"e_1_2_11_36_2","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.34509"},{"key":"e_1_2_11_37_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(16)00645-0"},{"key":"e_1_2_11_38_2","doi-asserted-by":"publisher","DOI":"10.1111\/pde.13600"}],"container-title":["Case Reports in Hematology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crh\/5825111","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1155\/crh\/5825111","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crh\/5825111","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,13]],"date-time":"2026-06-13T04:06:08Z","timestamp":1781323568000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1155\/crh\/5825111"}},"subtitle":[],"editor":[{"given":"Kalyani","family":"Sen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2026,1]]},"references-count":38,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"alternative-id":["10.1155\/crh\/5825111"],"URL":"https:\/\/doi.org\/10.1155\/crh\/5825111","archive":["Portico"],"relation":{},"ISSN":["2090-6560","2090-6579"],"issn-type":[{"value":"2090-6560","type":"print"},{"value":"2090-6579","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1]]},"assertion":[{"value":"2025-10-05","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-05-09","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-06-12","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"5825111"}}