ArticlePediatric blood & cancer2019
Castleman disease in pediatrics: Insights on presentation, treatment, and outcomes from a two-site retrospective cohort study.
Article in Pediatric blood & cancer, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers, 2 of them syntheses that pooled it.
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Who cites it
17 citing papers in PubMed, 2 syntheses or guidelines pooled it, 37 citations in OpenAlex.
- The clinical picture of Castleman disease: a systematic review and meta-analysis.Blood advances · 2024Pooled it
- Off-label use of canakinumab in pediatric rheumatology and rare diseases.Frontiers in medicine · 2022Pooled it
- Pediatric Idiopathic Multicentric Castleman Disease Is Often Severe But Responsive to Siltuximab.Pediatric blood & cancer · 2026Article
- Clinical features and therapeutic outcomes in pediatric patients with Castleman's disease: a retrospective cohort analysis.European journal of pediatrics · 2026Article
- Castleman Disease in Tunisia: A Multicenter Clinical, Prognostic, and Therapeutic Study.Advances in hematology · 2026Article
- Challenging diagnosis: unusual presentation of Unicentric Castleman disease-a case report.Oxford medical case reports · 2025Article
- Idiopathic multicentric Castleman disease in children: a single-center retrospective analysis.BMC pediatrics · 2025Article
- A hypothesis on treatment strategy of severe multicentric Castleman disease with continuous renal replacement therapy.Journal of cellular and molecular medicine · 2024Review
- Castleman Disease Presenting in the Neck: A Report of 3 Cases and a Literature Review.The American journal of case reports · 2024Review
- Clinical features and treatment outcomes of Castleman disease in children: a retrospective cohort in China.European journal of pediatrics · 2023Review
- Imaging evaluation of the pediatric mediastinum: new International Thymic Malignancy Interest Group classification system for children.Pediatric radiology · 2022Review
- Pediatric mediastinal masses.Pediatric radiology · 2022Review
- Transcriptome and unique cytokine microenvironment of Castleman disease.Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc · 2022Article
- Pediatric Lymphoid and Histiocytic Lesions in the Head and Neck.Head and neck pathology · 2021Review
- International evidence-based consensus diagnostic and treatment guidelines for unicentric Castleman disease.Blood advances · 2020Article
- Histologic and Laboratory Characteristics of Symptomatic and Asymptomatic Castleman Disease in the Pediatric Population.American journal of clinical pathology · 2020Article
- The French paediatric cohort of Castleman disease: a retrospective report of 23 patients.Orphanet journal of rare diseases · 2020Article
Corrections and comments
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Authors and funding
9 authors at 3 institutions in 1 country.
Funding
Abstract
backgroundCastleman disease (CD) is an uncommon lymphoproliferative disorder that is rare in pediatric populations; the literature describing this population is sparse. We sought to describe pediatric CD, including unicentric CD (UCD) and human herpes virus-8 (HHV8)-negative multicentric CD (MCD), in a multi-institutional cohort.
methodsWe retrospectively reviewed 24 patients, aged 0 to 26 years at diagnosis, who were diagnosed with CD between January 1, 2005, and May 16, 2017, at two tertiary children's hospitals. Demographic and clinical data were collected.
resultsMost patients (75%, 18/24) presented with UCD. All patients with MCD were HHV8-negative. The most common histopathologic variant was hyaline vascular (75%, 18/24). Plasma cell variant occurred in 33% (2/6 [95% confidence intervals (CI), 4-78%]) of patients with HHV8-negative MCD and 17% (3/18 [95% CI, 4-41%]) of patients with UCD. Systemic symptoms were present in 4 of 6 of patients with HHV8-negative MCD and 8 of 18 of patients with UCD. Anemia and laboratory inflammation occurred in both UCD and MCD patients, with nonsignificantly higher rates of anemia and elevated C-reactive protein in MCD patients. All but two UCD patients underwent gross total resection as definitive therapy. Among HHV8-negative MCD patients, a combination of resection, chemotherapy, and immunotherapy was used. No UCD patients and three of six HHV8-negative MCD patients experienced disease progression/relapse prior to lasting remission. There were no deaths.
conclusionPediatric patients with CD most commonly have unicentric, hyaline vascular variant disease. Pediatric patients with both UCD and MCD commonly have systemic inflammation and, despite risk of progression/relapse in MCD patients, ultimately have excellent survival.
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