Introduction: Typically, UCD presents with a lack of systemic symptoms, but may have some resulting from compression of nearby structures. MCD is rarely associated with vascular proliferations, but it has been proposed to result from elevated levels of vascular endothelial growth factor via IL-6 pathways. There have been published cases discussing hemangiomas in patients with MCD, however, cases of UCD with hemangiomas has not been reported.

Case Presentation: A 60-year-old African American male with past medical history of sarcoidosis, psoriasis, and unicentric castlemans disease presented in December 2022 for a follow-up for his palmoplantar psoriasis. From review of his medical history, this patient was diagnosed at the age of 57 with UCD. He presented with right neck lymphadenopathy and core needle biopsy revealed reactive lymphoid tissue. On examination in clinic, he had several asymptomatic discrete papules scattered on his trunk, face, and extremities.

PET demonstrated level V lymph node activity and he subsequently underwent neck dissection for lymph node removal. The formal diagnosis of UCD (hyaline vascular subtype) was made following clinicopathologic correlation. Afterwards, he was treated with rituximab 375mg weekly infusions x 4 due to concern for systemic symptoms and potential residual disease.

Follow up: At a follow-up appointment 2 months later, the patient was found to have multiple new lesions. Given the history of numerous biopsies with benign vascular proliferations, no treatment was advised at that time. This patient will continue to be closely monitored by dermatology and heme/onc,

Working Diagnosis: UCD with multiple discrete hemangiomas. While the diagnosis of UCD is maintained in this patient, he will continue to be followed closely for potential MCD.

References and Resources

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2. Fajgenbaum DC, Rosenbach M, van Rhee F, Nasir A, Reutter J. Eruptive Cherry Hemangiomatosis Associated With Multicentric Castleman Disease: A Case Report and Diagnostic Clue. JAMA Dermatol. 2013;149(2):204–208. doi:10.1001/jamadermatol.2013.1552
3. Gündüz E, Kırkızlar HO, Ümit EG, et al. Castleman Disease: A Multicenter Case Series from Turkey. Turk J Haematol. 2022;39(2):130-135. doi:10.4274/tjh.galenos.2022.2021.0670
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