J Korean Med Sci.  2004 Apr;19(2):311-314. 10.3346/jkms.2004.19.2.311.

Meningeal Relapse in a Patient with Acute Promyelocytic Leukemia: A Case Report and Review of the Literature

• Bae SH
• Ryoo HM
• Cho HS
• Lee JL
• Lee KH
• Hyun MS

• KMID: 1733498
• DOI: http://doi.org/10.3346/jkms.2004.19.2.311

Abstract

The involvement of central nervous system is rare in acute promyelocytic leukemia (APL). We report a APL patient of a 41 yr-old Korean male who presented with fever and petechia. Complete molecular remission was achieved with all-trans retinoic acid (ATRA), idarubicin, and cytarabine. Ten months later, he complained of a mild headache. The results of the physical examination and the complete blood counts were normal. The examination of cerebrospinal fluid showed the presence of promyelocyte. Bone marrow studies showed cytogenetic remission but with molecular relapse. He was treated with intrathecal and systemic chemotherapy.

Keyword

Leukemia, Promyelocytic, Acute; Central Nervous System; Recurrence

MeSH Terms

Adult
Granulocyte Precursor Cells
Human
Leukemia, Promyelocytic, Acute/*cerebrospinal fluid/*diagnosis
Male
Meninges
Recurrence

Figure

• 

  Fig. 1 Cells in cerebrospinal fluid with APL morphologic feature are noted (peroxidase stain, ×1,000).

Reference

1. Fenaux P, Le Deley MC, Castaigne S, Archimbaud E, Chomienne C, Link H, Guerci A, Duarte M, Daniel MT, Bowen D, et al. Effect of all-trans-retinoic acid in newly diagnosed acute promyelocytic leukemia. Result of a multicenter randomized trial. European APL 91 Group. Blood. 1993. 82:3241–3249.

2. Dekker AW, Elderson A, Punt K, Sixma JJ. Meningeal involvement in patients with acute nonlymphocytic leukemia. Incidence, management, and predictive factors. Cancer. 1985. 56:2078–2082.

3. Cassileth PA, Sylvester LS, Bennett JM, Begg CB. High peripheral blast count in adult acute myelogenous leukemia is a primary risk factor for CNS leukemia. J Clin Oncol. 1988. 6:495–498.

4. Wiernik PH, De Bellis R, Muxi P, Dutcher JP. Extramedullary acute promyelocytic leukemia. Cancer. 1996. 78:2510–2514.

5. Colovic N, Bogdanovic A, Miljic P, Jankovic G, Colovic M. Central nervous system relapse in acute promyelocytic leukemia. Am J Hematol. 2002. 71:60–61.

6. Patriarca F, Fili C, Geromin A, Sperotto A, Prosdocimo S, Fanin R. Activity of all-trans-retinoic acid in a case of central nervous system extramedullary relapse of acute promyelocytic leukemia. Eur J Haematol. 2002. 68:310–313.

7. Mathews V, Balasubramanian P, Shaji RV, George B, Chandy M, Srivastava A. Central nervous system relapse in a patient with acute promyelocytic leukaemia treated with arsenic tri-oxide. Br J Haematol. 2001. 113:257–258.

8. Evans GD, Grimwade DJ. Extramedullary disease in acute promyelocytic leukemia. Leuk Lymphoma. 1999. 33:219–229.

9. Au WY, Ma SK, Ooi C, Liang R, Kwong YL. Unusual manifestations of acute leukemia. Case 1. CNS extramedullary relapse of acute promyelocytic leukemia after arsenic trioxide-induced remission. J Clin Oncol. 2000. 18:3435–3437.

10. Anguita E, Villegas A, Gonzalez FA, Diaz-Mediavilla J, Lo Coco F. Acute promyelocytic leukemia relapse in the central nervous system during hematologic and molecular remission. Am J Hematol. 1999. 60:170–171.

11. Molero T, Valencia JM, Gomez-Casares MT. Central nervous system (CNS) infiltration in a case of promyelocytic leukemia. Haematologica. 1997. 82:637.

12. Mesa JR, Espinosa E, Losada R, Hernandez C, Martinez G, Hernandez P. Parotid and central nervous system relapse during complete hematologic remission in acute promyelocytic leukemia. Haematologica. 1999. 84:565–566.

13. Ko BS, Tang JL, Chen YC, Yao M, Wang CH, Shen MC, Tien HF. Extramedullary relapse after all-trans retinoic acid treatment in acute promyelocytic leukemia--the occurrence of retinoic acid syndrome is a risk factor. Leukemia. 1999. 13:1406–1408.

14. Liso V, Specchia G, Pogliani EM, Palumbo G, Mininni D, Rossi V, Teruzzi E, Mestice A, Coppi MR, Biondi A. Extramedullary involvement in patients with acute promyelocytic leukemia: a report of seven cases. Cancer. 1998. 83:1522–1528.

15. Martino B, Vincelli I, Marino A, Comis M, Ronco F, Nobile F. Meningeal relapse in a patient with acute promyelocytic leukaemia treated with all-trans retinoic acid. Br J Haematol. 1998. 100:606–607.

16. Lederman CA, Weisberger J, Seiter K, Feldman EJ. Differentiation of extramedullary acute promyelocytic leukemia by all-trans-retinoic acid. Leuk Lymphoma. 1995. 18:189–193.

17. Brown DM, Kimura AE, Ossoinig KC, Weiner GJ. Acute promyelocytic infiltration of the optic nerve treated by oral trans-retinoic acid. Ophthalmology. 1992. 99:1463–1467.

18. Kanakura Y, Yonezawa T, Hamaguchi Y, Otsuka A, Matayoshi Y, Kondoh H, Tamaki T, Katagiri S, Kanayama Y, Nishiura T, Aozasa K, Tarui S. Acute promyelocytic leukemia with an intracerebral mass and meningeal involvement after treatment of non-Hodgkin's lymphoma. Cancer. 1987. 59:94–98.

19. Solano Vercet C, Escudero A, Fernandez-Ranada JM. Meningeal relapse in acute promyelocytic leukaemia. Acta Haematol. 1983. 70:137–138.

20. Marra R, Stori S, Pagano L, Fioritoni G, Rabitti C, Sica S, Leone G, Torlontano G, Bizzi B. Central nervous system acute promyelocytic leukaemia: a report of three cases. Haematologia (Budap). 1989. 22:195–199.

21. Lugassy G, Halperin Y. Pituitary relapse of acute promyelocytic leukemia after ATRA. Int J Hematol. 1998. 68:101–102.

22. Currie J, Chee YL, Culligan DJ. Central nervous system relapse in acute promyelocytic leukaemia treated with ATRA. Br J Haematol. 1997. 99:469.

23. Pogliani EM, Fowst C, Marozzi A, Salvatore M, Polli EE. Cerebral parenchymal involvement in acute promyelocytic leukemia. A case report. Haematologica. 1988. 73:71–73.

24. Seldon M, Enno A. Central nervous system relapse in acute promyelocytic leukaemia treated with all-trans retinoic acid. Aust N Z J Med. 1998. 28:462.

25. Burry LD, Seki JT. CNS relapses of acute promyelocytic leukemia after all-trans retinoic acid. Ann Pharmacother. 2002. 36:1900–1906.

26. Marchetti M, Falanga A, Giovanelli S, Oldani E, Barbui T. All-trans-retinoic acid increases adhesion to endothelium of the human promyelocytic leukaemia cell line NB4. Br J Haematol. 1996. 93:360–366.

27. Saiki I, Fujii H, Yoneda J, Abe F, Nakajima M, Tsuruo T, Azuma I. Role of aminopeptidase N (CD13) in tumor-cell invasion and extracellular matrix degradation. Int J Cancer. 1993. 54:137–143.

28. Specchia G, Lo Coco F, Vignetti M, Avvisati G, Fazi P, Albano F, Di Raimondo F, Martino B, Ferrara F, Selleri C, Liso V, Mandelli F. Extramedullary involvement at relapse in acute promyelocytic leukemia patients treated or not with all-trans retinoic acid: a report by the Gruppo Italiano Malattie Ematologiche dell'Adulto. J Clin Oncol. 2001. 19:4023–4028.

29. Soignet SL, Frankel SR, Douer D, Tallman MS, Kantarjian H, Calleja E, Stone RM, Kalaycio M, Scheinberg DA, Steinherz P, Sievers EL, Coutre S, Dahlberg S, Ellison R, Warrell RP Jr. United States multicenter study of arsenic trioxide in relapsed acute promyelocytic leukemia. J Clin Oncol. 2001. 19:3852–3860.