Volume 8, Issue 4, December 2020, Page: 100-103
Castleman Disease in Children from Histopathology to Therapy
Asmaa Hamoda, Department of Pediatric Oncology, Children Cancer Hospital Egypt, Cairo, Egypt; Department of Pediatric Oncology, National Cancer Institute, Cairo University, Cairo, Egypt
Hanaa Rashad, Department of Pediatric Oncology, Children Cancer Hospital Egypt, Cairo, Egypt
Ola Ahmad, Department of Clinical Research, Children Cancer Hospital Egypt, Cairo, Egypt
Hala Reda, Department of Clinical Pathology, Children Cancer Hospital Egypt, Cairo, Egypt; Department of Clinical Pathology, National Cancer Institute, Cairo University, Cairo, Egypt
Iman Zaki, Department of Radiodiagnosis, National Cancer Institute, Cairo University, Cairo, Egypt; Department of Radiodiagnosis, Children Cancer Hospital Egypt, Cairo, Egypt
Naglaa Elkinaai, Department of Pathology, National Cancer Institute, Cairo University, Cairo, Egypt; Department of Pathology, Children Cancer Hospital Egypt, Cairo, Egypt
Mohamed Sedki, Department of Pediatric Oncology, Children Cancer Hospital Egypt, Cairo, Egypt; Department of Pediatrics, National Research Center, Cairo, Egypt
Alaa El Hadad, Department of Pediatric Oncology, Children Cancer Hospital Egypt, Cairo, Egypt; Department of Pediatric Oncology, National Cancer Institute, Cairo University, Cairo, Egypt
Samah Semary, Department of Pediatric Oncology, Children Cancer Hospital Egypt, Cairo, Egypt; Department of Clinical Oncology, Beni-suef University, Beni-suef, Egypt
Received: Apr. 11, 2020;       Accepted: Dec. 15, 2020;       Published: Dec. 22, 2020
DOI: 10.11648/j.crj.20200804.16       View        Downloads  
Abstract
Background: Castleman disease (CD) describes a group of rare lymphoproliferative disorder with characteristic histopathology. It presents with heterogeneous clinical features whether unicentric (UC) or multicentric disease (MCD). The aim of the work was to describe clinic-pathological characteristics, management and outcome of different types of castleman disease in CCHE. It is a retrospective study, all children with (CD) treated from July 2007 till end of 2017 were analyzed as regard diagnosis, management and outcome. The results showed that, twelve patients with a median age of 11.5 years (ranging from 4 - to 17 years) were enrolled. Eight of them (66.6%) were males and 4 (33.3%) were females (33.3%). Histopathology was either hyaline vascular in 8 patients (66.6%) or plasma cell variant in 4 patients (33.3%). Nine patients (75%) had uni-centric disease (UC) with lymphadenopathy. Three patients (25%) were multi-centric disease (MCD); out of them one patient had immune bi-cytopenia, small intestinal thickening and splenomegaly and another one had extensive pulmonary involvement and respiratory distress, while the third patient had pleural effusion and ascites. HIV antibody was negative for all patients. Regarding the three MCD patients, IL-6 was normal in one patient, and extremely high in second one, while it hasn’t been done for the third patient. All UC patients underwent surgical excision and are alive with no active disease till now. All MCD patients received systemic steroids as initial therapy, followed by R- CHOP (Rituximab-cyclophosphamide-doxorubicin-vincristine-prednisone) as salvage therapy, except for one patient with pulmonary involvement who didn’t achieved clinical response and received single agent Rituximab with Anti IL-6. Unfortunately this patient is still having uncontrolled systemic manifestation, while the other two patients showed partial response. In conclusion, Uni-centric castleman disease is a localized surgically cured disease. MCD treatment remains challenging, and the outcome is controversial, so Uniform treatment guidelines are mandatory.
Keywords
Castleman Disease, Unicentric Disease, Multicentric Disease, Histopathology, Ritoxomab, Anti-IL6
To cite this article
Asmaa Hamoda, Hanaa Rashad, Ola Ahmad, Hala Reda, Iman Zaki, Naglaa Elkinaai, Mohamed Sedki, Alaa El Hadad, Samah Semary, Castleman Disease in Children from Histopathology to Therapy, Cancer Research Journal. Vol. 8, No. 4, 2020, pp. 100-103. doi: 10.11648/j.crj.20200804.16
Copyright
Copyright © 2020 Authors retain the copyright of this article.
This article is an open access article distributed under the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Reference
[1]
Fajgenbaum dc, van rhee f, nabel cs. Hhv-8-negative, idiopathic multicentric castleman disease: novel insights into biology, Pathogenesis, and therapy. Blood. 2014; 123 (19): 2924-2933.
[2]
Liu AY, nabel CS, finkelman BS, et al. Idiopathic multicentricCastleman’s disease: a systematic literature review. Lancet haematol. 2016; 3 (4): e163-e175.
[3]
Munshi N, mehra M, van de velde H, desai A, potluri R, vermeulen J. Use of a claims database to characterize and estimate the incidence rateFor castleman disease. Leuk lymphoma. 2015; 56 (5): 1252-1260.
[4]
Piero Farruggia, 1 Antonino Trizzino, 1 Nunzia Scibetta, 2 et al, Castleman's disease in childhood: report of three cases and review of the literature, Ital J Pediatr. 2011; 37: 50.
[5]
V. I. Burlakov, A. L. Kozlova, A. J. Shcherbina, et al, THU0587 Clinical Characterization of Castleman's Disease in A Group of Pediatric Patients, Poster Presentations 2016
[6]
Seth J. Kligerman, Aaron Auerbach, Teri J. Franks, Castleman Disease of the Thorax: Clinical, Radiologic, and Pathologic Correlation: From the Radiologic Pathology Archives, RadioGraphicsVol. 36, No. 5, 2016.
[7]
Weisenburger DD, Nathwani BN, Winberg CD, Rappaport H. Multicentric angiofollicular lymph node hyperplasia: a clinicopathologic study of 16 cases. Hum Pathol. 1985; 16: 162–72. doi: 10.1016/S0046-8177(85)80065-4.
[8]
Frizzera G, Peterson BA, Bayrd ED, Goldman A. A systemic lymphoproliferative disorder with morphologic features of Castleman's disease: clinical findings and clinicopathologic correlations in 15 patients. J Clin Oncol. 1985; 3 (9): 1202–16.
[9]
Kasantikul V, Panyavoravut V, Benjavongkulchai S, Panichabhongse V. Castleman's disease: a clinicopathologic study of 12 cases. J Med Assoc Thai. 1997; 80 (3): 195–201.
[10]
Sarra Benmiloud, Sana Chaouki, Samir Atmani, et al, Multicentric Castleman’s Disease in a Child Revealed by Chronic Diarrhea, Case Reports in Pediatrics, Volume 2015, Article ID 689206, 4 pages.
[11]
David C. Fajgenbaum, Thomas S. Uldrick, Adam Bagg, et al, International, evidence-based consensus diagnostic criteria for HHV-8–negative/idiopathic multicentric Castleman disease, Blood 2017 129: 1646-1657; doi: https://doi.org/10.1182/blood-2016-10-746933.
[12]
Brandt SJ, Bodine DM, Dunbar CE, Nienhuis AW. Dysregulated interleukin expression produces a syndrome resembling Castleman's disease in mice. J Clin Invest. 1990; 86 (2): 592–9. doi: 10.1172/JCI114749.
[13]
Ye B, Gao SG, Li W et al. A retrospective study of unicentric and multicentric Castleman’s disease: a report of 52 patients. Med Oncol 2010; 27 (4): 1171–1178.
[14]
Talat N, Belgaumkar AP, Schulte KM. Surgery in Castleman’s disease: a systematic review of 404 published cases. Ann Surg 2012; 255 (4): 677–684.
[15]
Summerfield GP, Taylor W, Bellingham AJ, Goldsmith HJ. Hyaline-vascular variant of angiofollicular lymph node hyperplasia with systemic manifestations and response to corticosteroids. J Clin Pathol. 1983; 36 (9): 1005–11. doi: 10.1136/jcp.36.9.1005.
[16]
Bower M, Veraitch O, Szydlo R, Charles P, Kelleher P, Gazzard B, Nelson M, Stebbing J. Cytokine changes during rituximab therapy in HIV-associated multicentric Castleman disease. Blood. 2009; 113 (19): 4521–4. doi: 10.1182/blood-2008-12-197053.
[17]
David Green, MD, PhD reviewing Van Rhee F et al, Treatment Guidelines for Idiopathic Multicentric Castleman Disease, Blood 2018.
Browse journals by subject