Abstract
Data on clinical features and outcome in pediatric follicular lymphoma (pFL) are scarce. The aim of this retrospective study including 13 EICNHL and/or i-BFM study group members was to assess clinical characteristics and course in a series of 63 pFL patients. pFL was found to be associated with male gender (3:1), older age (72 % ≥10 years old), low serum LDH levels (<500 U/l in 75 %), grade 3 histology (in 88 %), and limited disease (87 % stage I/II disease), mostly involving the peripheral lymph nodes. Forty-four out of sixty-three patients received any polychemotherapy and 1/63 rituximab only, while 17/63 underwent a “watch and wait” strategy. Of 36 stage I patients, 30 had complete resections. Only one patient relapsed; 2-year event-free survival and overall survival were 94 ± 5 and 100 %, respectively, after a median follow-up of 2.2 years. Conclusively, treatment outcome in pFL seems to be excellent with risk-adapted chemotherapy or after complete resection and an observational strategy only.
Similar content being viewed by others
References
Agrawal R, Wang J (2009) Pediatric follicular lymphoma: a rare clinicopathologic entity. Arch Pathol Lab Med 133:142–146
Anderson JR, Armitage JO, Weisenburger DD (1998) Epidemiology of the non-Hodgkin's lymphomas: distributions of the major subtypes differ by geographic locations. Non-Hodgkin's lymphoma classification project. Ann Oncol 9:717–720
Oschlies I, Salaverria I, Mahn F, Meinhardt A, Zimmermann M, Woessmann W, Burkhardt B, Gesk S, Krams M, Reiter A, Siebert R, Klapper W (2010) Pediatric follicular lymphoma—a clinicopathological study of a population-based series of patients treated within the non-Hodgkin's lymphoma-Berlin–Frankfurt–Munster (NHL-BFM) multicenter trials. Haematologica 95:253–259
Kansal R, Singleton TP, Ross CW, Finn WG, Padmore RF, Schnitzer B (2002) Follicular Hodgkin lymphoma: a histopathologic study. Am J Clin Pathol 117:29–35
Louissaint A Jr, Ackerman AM, Dias-Santagata D, Ferry JA, Hochberg EP, Huang MS, Iafrate AJ, Lara DO, Pinkus GS, Salaverria I, Siddiquee Z, Siebert R, Weinstein HJ, Zukerberg LR, Harris NL, Hasserjian RP (2012) Pediatric-type nodal follicular lymphoma: an indolent clonal proliferation in children and adults with high proliferation index and no BCL2 rearrangement. Blood 120:2395–2404
Atra A, Meller ST, Stevens RS, Hobson R, Grundy R, Carter RL, Pinkerton CR (1998) Conservative management of follicular non-Hodgkin's lymphoma in childhood. Br J Haematol 103:220–223
Kumar R, Galardy PJ, Dogan A, Rodriguez V, Khan SP (2011) Rituximab in combination with multiagent chemotherapy for pediatric follicular lymphoma. Pediatr Blood Cancer 57:317–320
Finn LS, Viswanatha DS, Belasco JB, Snyder H, Huebner D, Sorbara L, Raffeld M, Jaffe ES, Salhany KE (1999) Primary follicular lymphoma of the testis in childhood. Cancer 85:1626–1635
Lones MA, Raphael M, McCarthy K, Wotherspoon A, Terrier-Lacombe MJ, Ramsay AD, Maclennan K, Cairo MS, Gerrard M, Michon J, Patte C, Pinkerton R, Sender L, Auperin A, Sposto R, Weston C, Heerema NA, Sanger WG, von Allmen D, Perkins SL (2012) Primary follicular lymphoma of the testis in children and adolescents. J Pediatr Hematol Oncol 34:68–71
Lorsbach RB, Shay-Seymore D, Moore J, Banks PM, Hasserjian RP, Sandlund JT, Behm FG (2002) Clinicopathologic analysis of follicular lymphoma occurring in children. Blood 99:1959–1964
McNamara C, Davies J, Dyer M, Hoskin P, Illidge T, Lyttelton M, Marcus R, Montoto S, Ramsay A, Wong WL, Ardeshna K (2012) Guidelines on the investigation and management of follicular lymphoma. Br J Haematol 156:446–467
Pinto A, Hutchison RE, Grant LH, Trevenen CL, Berard CW (1990) Follicular lymphomas in pediatric patients. Mod Pathol 3:308–313
Murphy SB, Fairclough DL, Hutchison RE, Berard CW (1989) Non-Hodgkin's lymphomas of childhood: an analysis of the histology, staging, and response to treatment of 338 cases at a single institution. J Clin Oncol 7:186–193
Amos Burke GA, Imeson J, Hobson R, Gerrard M (2003) Localized non-Hodgkin's lymphoma with B cell histology: cure without cyclophosphamide? A report of the United Kingdom Children's Cancer Study Group on studies NHL 8501 and NHL 9001 (1985–1996). Br J Haematol 121:586–591
Fujita N, Kobayashi R, Takimoto T, Nakagawa A, Ueda K, Horibe K (2011) Results of the Japan Association of Childhood Leukemia Study (JACLS) NHL-98 protocol for the treatment of B cell non-Hodgkin lymphoma and mature B cell acute lymphoblastic leukemia in childhood. Leuk Lymphoma 52:223–229
Gerrard M, Cairo MS, Weston C, Auperin A, Pinkerton R, Lambilliote A, Sposto R, McCarthy K, Lacombe MJ, Perkins SL, Patte C (2008) Excellent survival following two courses of COPAD chemotherapy in children and adolescents with resected localized B cell non-Hodgkin's lymphoma: results of the FAB/LMB 96 international study. Br J Haematol 141:840–847
Pillon M, Di Tullio MT, Garaventa A, Cesaro S, Putti MC, Favre C, Lippi A, Surico G, Di Cataldo A, D'Amore E, Zanesco L, Rosolen A (2004) Long-term results of the first Italian Association of Pediatric Hematology and Oncology protocol for the treatment of pediatric B cell non-Hodgkin lymphoma (AIEOP LNH92). Cancer 101:385–394
Woessmann W, Seidemann K, Mann G, Zimmermann M, Burkhardt B, Oschlies I, Ludwig WD, Klingebiel T, Graf N, Gruhn B, Juergens H, Niggli F, Parwaresch R, Gadner H, Riehm H, Schrappe M, Reiter A (2005) The impact of the methotrexate administration schedule and dose in the treatment of children and adolescents with B cell neoplasms: a report of the BFM Group Study NHL-BFM95. Blood 105:948–958
Murphy SB (1980) Classification, staging, and end results of treatment of childhood non-Hodgkin's lymphomas: dissimilarities from lymphomas in adults. Semin Oncol 7:332–339
Kridel R, Sehn LH, Gascoyne RD (2012) Pathogenesis of follicular lymphoma. J Clin Invest 122:3424–3431
Marcus R, Imrie K, Solal-Celigny P, Catalano JV, Dmoszynska A, Raposo JC, Offner FC, Gomez-Codina J, Belch A, Cunningham D, Wassner-Fritsch E, Stein G (2008) Phase III study of R-CVP compared with cyclophosphamide, vincristine, and prednisone alone in patients with previously untreated advanced follicular lymphoma. J Clin Oncol 26:4579–4586
Perkins SL, Gross TG (2011) Pediatric indolent lymphoma–would less be better? Pediatr Blood Cancer 57:189–190
Goldman S, Smith L, Anderson JR, Perkins S, Harrison L, Geyer MB, Gross TG, Weinstein H, Bergeron S, Shiramizu B, Sanger W, Barth M, Zhi J, Cairo MS (2012) Rituximab and FAB/LMB 96 chemotherapy in children with Stage III/IV B-cell non-Hodgkin lymphoma: a Children's Oncology Group report. Leukemia
Shankar A, Hall GW, Gorde-Grosjean S, Hasenclever D, Leblanc T, Hayward J, Lambilliotte A, Daw S, Perel Y, McCarthy K, Lejars O, Coulomb A, Oberlin WO, Wallace WH, Landman-Parker J (2012) Treatment outcome after low intensity chemotherapy (CVP) in children and adolescents with early stage nodular lymphocyte predominant Hodgkin's lymphoma—an Anglo–French collaborative report. Eur J Cancer 48:1700–1706
Marcus R, Imrie K, Belch A, Cunningham D, Flores E, Catalano J, Solal-Celigny P, Offner F, Walewski J, Raposo J, Jack A, Smith P (2005) CVP chemotherapy plus rituximab compared with CVP as first-line treatment for advanced follicular lymphoma. Blood 105:1417–1423
Meinhardt A, Burkhardt B, Zimmermann M, Borkhardt A, Kontny U, Klingebiel T, Berthold F, Janka-Schaub G, Klein C, Kabickova E, Klapper W, Attarbaschi A, Schrappe M, Reiter A (2010) Phase II window study on rituximab in newly diagnosed pediatric mature B cell non-Hodgkin's lymphoma and Burkitt leukemia. J Clin Oncol 28:3115–3121
Acknowledgments
We thank all participating institutions and physicians for their support of the study. This EICNHL and i-BFM paper was written on behalf of the Berlin–Frankfurt–Münster (BFM) study group (Austria, Germany, Switzerland, Czech Republic), Associazione Italiana Ematologia Oncologia Pediatrica (AIEOP), UK Children's Cancer and Leukemia study group (CCLG), Nordic Society of Pediatric Hematology and Oncology (NOPHO), Belgian Society of Pediatric Hematology and Oncology, Dutch Childhood Oncology Group (DCOG), Hungarian Pediatric Oncology Network, Japanese Pediatric Leukemia/Lymphoma study group (JPLSG), and Hong Kong Pediatric Hematology and Oncology study group (HKPHOSG). This work was supported by the Cancer Research UK, the Forschungshilfe Station Peiper (BFM Germany), the St. Anna Kinderkrebsforschung (BFM Austria), the Czech Ministry of Health supported projects for conceptual development of research organization 00064203 and 65269705 (BFM Czech Republic), the Associazione Italiana Contro le Leucemie and Fondazione Citta della Speranza (AIEOP), and the Ministry of Health, Labor, and Welfare of Japan (JPLSG).
Conflicts of interest
The authors declare no competing financial interests.
Author information
Authors and Affiliations
Consortia
Corresponding author
Rights and permissions
About this article
Cite this article
Attarbaschi, A., Beishuizen, A., Mann, G. et al. Children and adolescents with follicular lymphoma have an excellent prognosis with either limited chemotherapy or with a “watch and wait” strategy after complete resection. Ann Hematol 92, 1537–1541 (2013). https://doi.org/10.1007/s00277-013-1785-2
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00277-013-1785-2