A case report of cutaneous Langerhans cell Histiocytosis in an infant: a therapeutic challenge.

IF 0.4 Q3 MEDICINE, GENERAL & INTERNAL
Oxford Medical Case Reports Pub Date : 2025-07-27 eCollection Date: 2025-07-01 DOI:10.1093/omcr/omaf112
Linda Khanje, Khaled Khanje, Shahd Alqato
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Abstract

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.

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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。
朗格汉斯细胞组织细胞增生症(LCH)是一种罕见的未成熟cd1a阳性树突状细胞的克隆性疾病,最常见于儿童,发病率在1至3岁之间达到高峰。本报告提出一个独特的病例多局灶性皮肤LCH与颈部淋巴结病在一个7个月大的婴儿。患者表现出一种不寻常的治疗抵抗模式,在使用甲氨蝶呤获得缓解之前,对皮质类固醇和长春花碱-强的松没有充分的反应。这突出了治疗难治性儿童LCH的挑战,以及在这种情况下需要替代治疗方法。这个病例强调了早期诊断的重要性,量身定制的治疗方案,以及甲氨蝶呤在难治性病例中的潜在效用。综合评估和多学科护理对于控制这种罕见疾病和尽量减少其对生活质量的影响至关重要。
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来源期刊
Oxford Medical Case Reports
Oxford Medical Case Reports MEDICINE, GENERAL & INTERNAL-
CiteScore
0.90
自引率
0.00%
发文量
125
审稿时长
19 weeks
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.
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