ArticleFrontiers in medicine2026
Diagnostic delay and histopathological subtyping challenges in idiopathic multicentric Castleman disease, not otherwise specified: a case report of a three-hospital odyssey.
Article in Frontiers in medicine, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Background: Castleman disease (CD) is a group of rare and heterogeneous lymphoproliferative disorders. Accurate diagnosis and subtyping remain challenging. This report describes a rare case of idiopathic multicentric Castleman disease, not otherwise specified (iMCD-NOS), highlighting the causes of misdiagnosis, diagnostic strategy, and critical features of clinical subtyping. Case presentation: A 53-year-old male presented with a 4-month history of persistent right submandibular lymphadenopathy. During this period, he experienced two febrile episodes (max temp 39.0 °C), each lasting 2 days, without associated respiratory symptoms. The fever resolved following intravenous antibiotic therapy at a local hospital, however, the lymphadenopathy did not improve and progressively enlarged. Core needle biopsy of the right submandibular lymph node at another municipal hospital revealed acute and chronic inflammation with fibrosis. He also reported occasional low-grade fever and night sweats, prompting admission to our hospital for further evaluation. On admission, physical examination revealed the largest lymph node measuring approximately 2.5 × 2.0 cm and purplish-red papules on the bilateral lower extremities. Laboratory findings revealed leukocytosis, eosinophilia, platelet (PLT) count: 296 × 10 Conclusion: This case illustrates the diagnostic challenges of idiopathic multicentric Castleman disease-not otherwise specified (iMCD-NOS) and underscores the importance of excisional biopsy over core needle biopsy, systematic exclusion of differential diagnoses, and multidisciplinary collaboration. The patient's journey through three hospitals before diagnosis offers three key lessons for clinicians: (1) The possibility of Castleman disease should be considered when a triad of unexplained lymphadenopathy, systemic symptoms, and elevated inflammatory markers is present. (2) When multicentric lymphadenopathy coexists with systemic inflammation, excisional biopsy-rather than core needle biopsy-is essential for an accurate diagnosis. (3) Systematic exclusion of HHV-8 infection, TAFRO/IPL features, and other mimickers is mandatory for proper iMCD-NOS subtyping. Enhanced awareness of this disease, as provided by this report, may facilitate early recognition and reduce diagnostic delays.
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