{"title":"婴儿皮肤朗格汉斯细胞组织细胞增多症一例报告:一个治疗挑战。","authors":"Linda Khanje, Khaled Khanje, Shahd Alqato","doi":"10.1093/omcr/omaf112","DOIUrl":null,"url":null,"abstract":"<p><p>Langerhans Cell Histiocytosis (LCH) is a rare clonal disorder of immature CD1a-positive dendritic cells, most commonly affecting children, with peak incidence between ages 1 and 3. This report presents a unique case of multifocal cutaneous LCH with cervical lymphadenopathy in a 7-month-old infant. The patient exhibited an unusual pattern of treatment resistance, failing to respond adequately to corticosteroids and vinblastine-prednisone, before achieving remission with methotrexate. This highlights the therapeutic challenges in managing refractory pediatric LCH and the need for alternative treatment approaches in such cases. This case emphasizes the importance of early diagnosis, tailored treatment regimens, and the potential utility of methotrexate in refractory cases. Comprehensive evaluation and multidisciplinary care are critical for managing this rare disease and minimizing its impact on quality of life.</p>","PeriodicalId":45318,"journal":{"name":"Oxford Medical Case Reports","volume":"2025 7","pages":"omaf112"},"PeriodicalIF":0.4000,"publicationDate":"2025-07-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12296381/pdf/","citationCount":"0","resultStr":"{\"title\":\"A case report of cutaneous Langerhans cell Histiocytosis in an infant: a therapeutic challenge.\",\"authors\":\"Linda Khanje, Khaled Khanje, Shahd Alqato\",\"doi\":\"10.1093/omcr/omaf112\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Langerhans Cell Histiocytosis (LCH) is a rare clonal disorder of immature CD1a-positive dendritic cells, most commonly affecting children, with peak incidence between ages 1 and 3. This report presents a unique case of multifocal cutaneous LCH with cervical lymphadenopathy in a 7-month-old infant. The patient exhibited an unusual pattern of treatment resistance, failing to respond adequately to corticosteroids and vinblastine-prednisone, before achieving remission with methotrexate. This highlights the therapeutic challenges in managing refractory pediatric LCH and the need for alternative treatment approaches in such cases. This case emphasizes the importance of early diagnosis, tailored treatment regimens, and the potential utility of methotrexate in refractory cases. Comprehensive evaluation and multidisciplinary care are critical for managing this rare disease and minimizing its impact on quality of life.</p>\",\"PeriodicalId\":45318,\"journal\":{\"name\":\"Oxford Medical Case Reports\",\"volume\":\"2025 7\",\"pages\":\"omaf112\"},\"PeriodicalIF\":0.4000,\"publicationDate\":\"2025-07-27\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12296381/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Oxford Medical Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1093/omcr/omaf112\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2025/7/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q3\",\"JCRName\":\"MEDICINE, GENERAL & INTERNAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Oxford Medical Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1093/omcr/omaf112","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/7/1 0:00:00","PubModel":"eCollection","JCR":"Q3","JCRName":"MEDICINE, GENERAL & INTERNAL","Score":null,"Total":0}
A case report of cutaneous Langerhans cell Histiocytosis in an infant: a therapeutic challenge.
Langerhans Cell Histiocytosis (LCH) is a rare clonal disorder of immature CD1a-positive dendritic cells, most commonly affecting children, with peak incidence between ages 1 and 3. This report presents a unique case of multifocal cutaneous LCH with cervical lymphadenopathy in a 7-month-old infant. The patient exhibited an unusual pattern of treatment resistance, failing to respond adequately to corticosteroids and vinblastine-prednisone, before achieving remission with methotrexate. This highlights the therapeutic challenges in managing refractory pediatric LCH and the need for alternative treatment approaches in such cases. This case emphasizes the importance of early diagnosis, tailored treatment regimens, and the potential utility of methotrexate in refractory cases. Comprehensive evaluation and multidisciplinary care are critical for managing this rare disease and minimizing its impact on quality of life.
期刊介绍:
Oxford Medical Case Reports (OMCR) is an open access, peer-reviewed online journal publishing original and educationally valuable case reports that expand the field of medicine. The journal covers all medical specialities including cardiology, rheumatology, nephrology, oncology, neurology, and reproduction, comprising a comprehensive resource for physicians in all fields and at all stages of training. Oxford Medical Case Reports deposits all articles in PubMed Central (PMC). Physicians and researchers can find your work through PubMed , helping you reach the widest possible audience. The journal is also indexed in the Web of Science Core Collection . Oxford Medical Case Reports publishes case reports under the following categories: Allergy Audiovestibular medicine Cardiology and cardiovascular systems Critical care medicine Dermatology Emergency medicine Endocrinology and metabolism Gastroenterology and hepatology Geriatrics and gerontology Haematology Immunology Infectious diseases and tropical medicine Medical disorders in pregnancy Medical ophthalmology Nephrology Neurology Oncology Paediatrics Pain Palliative medicine Pharmacology and pharmacy Psychiatry Radiology, nuclear medicine, and medical imaging Respiratory disorders Rheumatology Sexual and reproductive health Sports Medicine Substance abuse.