Castleman disease in a pediatric liver transplant recipient: a case report and literature review.

Publisher: Munksgaard Country of Publication: Denmark NLM ID: 9802574 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1399-3046 (Electronic) Linking ISSN: 13973142 NLM ISO Abbreviation: Pediatr Transplant Subsets: MEDLINE

Original Publication: Copenhagen ; Malden, MA : Munksgaard, c1997-

Castleman Disease/*complications ; Liver Failure/*complications ; Liver Failure/*therapy ; Liver Transplantation/*methods; Adolescent ; Adult ; Antigens, CD20/biosynthesis ; B-Lymphocytes/metabolism ; CD3 Complex/biosynthesis ; Castleman Disease/diagnosis ; Graft Survival ; Herpesvirus 8, Human/genetics ; Humans ; Immunosuppressive Agents/therapeutic use ; Infant, Newborn ; Liver Transplantation/adverse effects ; Lymphatic Diseases/diagnosis ; Male ; Middle Aged ; Polymerase Chain Reaction ; Sirolimus/administration & dosage ; Splenomegaly/diagnosis ; T-Lymphocytes/metabolism ; Tacrolimus/administration & dosage ; Tomography, X-Ray Computed/methods

Castleman disease is a rare hematologic disorder, closely linked to the HHV-8, and most commonly observed in immunocompromised individuals. Thirteen months following a liver transplant for CPS-1 defect, a 15-month-old boy presented with fevers, anemia, and growth retardation. Abdominal CT scan showed splenomegaly and generalized lymphadenopathy. Histology of chest wall lymph nodes revealed a mixed CD3+ T-cell and CD20+ B-cell population with atretic germinal centers consistent with multicentric Castleman disease. Qualitative DNA PCR detected HHV-8 in the resected lymph node and in the blood, supporting the diagnosis. Immunosuppression was tapered, and he was transitioned from tacrolimus to sirolimus. His graft function remained stable, and repeat imaging showed regression of the lymphadenopathy. The child is living one yr after Castleman disease diagnosis with a well-functioning graft. Castleman disease is a potential complication of solid organ transplant and HHV-8 infection. Reduction in immunosuppression and switch to sirolimus may be an effective strategy to treat this condition.
(© 2011 John Wiley & Sons A/S.)

0 (Antigens, CD20)
0 (CD3 Complex)
0 (Immunosuppressive Agents)
W36ZG6FT64 (Sirolimus)
WM0HAQ4WNM (Tacrolimus)

Date Created: 20111029 Date Completed: 20130110 Latest Revision: 20171116

20240829

10.1111/j.1399-3046.2011.01570.x

22032720