Korean J Lab Med.  2011 Oct;31(4):225-230. 10.3343/kjlm.2011.31.4.225.

Myelomatous Pleural Effusion: A Case Series in a Single Institution and Literature Review

• Cho YU
• Chi HS
• Park CJ
• Jang S
• Seo EJ
• Suh C

• KMID: 1781684
• DOI: http://doi.org/10.3343/kjlm.2011.31.4.225

Abstract

BACKGROUND
Myelomatous pleural effusion (MPE) is rare in myeloma patients. We present a consecutive series of patients with MPE in a single institution.
METHODS
We retrospectively reviewed the medical records of 19 patients diagnosed with MPE between 1989 and 2008 at the Asan Medical Center. Diagnoses were confirmed by cytologic identification of malignant plasma cells in the pleural fluid.
RESULTS
Our patients showed dominance of IgA (36.8%) and IgD (31.6%) subtypes. Of 734 myeloma patients, the incidence of MPE was remarkably high for the IgD myeloma subtype (16.7%), compared to the other subtypes (1.4% for IgG and 4.6% for IgA). At the time of diagnosis of MPE, elevated serum beta2-microglobulin, anemia, elevated serum lactate dehydrogenase, and elevated creatinine levels were found in 100%, 89.5%, 83.3%, and 57.9% of the patients, respectively. Approximately one-third (31.3%) of the patients had adenosine deaminase (ADA) activities in their pleural fluid exceeding the upper limit of the reported cutoff values for tuberculous pleural effusion (55.8 U/L). Chromosome 13 abnormality was seen in 77.8% of the tested patients. The median survival period from the development of MPE was 2.8 months.
CONCLUSIONS
Patients with MPE have aggressive clinical and laboratory characteristics. The preponderance of IgD myeloma in MPE patients is a noteworthy finding because IgD myeloma is a rare subtype. Elevated ADA activity in the pleural fluid is also noteworthy, and may be helpful for detecting MPE. Physicians treating myeloma patients should monitor the development of MPE and consider the possibility of a worse clinical course.

Keyword

Myelomatous pleural effusion; IgD myeloma; Adenosine deaminase; Chromosome 13 abnormality

MeSH Terms

Adenosine Deaminase/metabolism
Adult
Aged
Chromosomes, Human, Pair 13
Creatine/blood
Diagnosis, Differential
Female
Humans
Immunoglobulin A/metabolism
Immunoglobulin D/metabolism
L-Lactate Dehydrogenase/blood
Male
Middle Aged
Multiple Myeloma/diagnosis
Plasma Cells/pathology
Pleural Effusion, Malignant/*diagnosis/mortality/pathology
Retrospective Studies
Survival Rate
beta 2-Microglobulin/blood

Figure

• 

  Fig. 1 Malignant plasma cells in the pleural effusion (case M). The cells have large, eccentrically placed and pleomorphic nuclei and prominent nucleoli (Wright-Giemsa stain, ×1,000).

Reference

1. Kintzer JS, Rosenow EC, Kyle RA. Thoracic and pulmonary abnormalities in multiple myeloma. A review of 958 cases. Arch Intern Med. 1978; 138:727–730. PMID: 646535.

2. Alexandrakis MG, Passam FH, Kyriakou DS, Bouros D. Pleural effusions in hematologic malignancies. Chest. 2004; 125:1546–1555. PMID: 15078773.

3. Rodriguez JN, Pereira A, Martinez JC, Conde J, Pujol E. Pleural effusion in multiple myeloma. Chest. 1994; 105:622–624. PMID: 8306782.

4. Meoli A, Willsie S, Fiorella R. Myelomatous pleural effusion. South Med J. 1997; 90:65–68. PMID: 9003828.

5. Kim YJ, Kim SJ, Min K, Kim HY, Kim HJ, Lee YK, et al. Multiple myeloma with myelomatous pleural effusion: a case report and review of literature. Acta Haematol. 2008; 120:108–111. PMID: 18957845.

6. Kamble R, Wilson CS, Fassas A, Desikan R, Siegel DS, Tricot G, et al. Malignant pleural effusion of multiple myeloma: prognostic factors and outcome. Leuk Lymphoma. 2005; 46:1137–1142. PMID: 16085553.

7. International Myeloma Working Group. Criteria for the classification of monoclonal gammopathies, multiple myeloma and related disorders: a report of the International Myeloma Working Group. Br J Haematol. 2003; 121:749–757. PMID: 12780789.

8. Munshi NC, Longo DL, Anderson KC. Fauci AS, Braunwald E, Kasper DL, editors. Plasma cell disorders. Harrison's principles of internal medicine. 2008. 17th ed. New York, USA: McGraw Hill Medical;p. 700–707.

9. Shimamoto Y, Anami Y, Yamaguchi M. A new risk grouping for IgD myeloma based on analysis of 165 Japanese patients. Eur J Haematol. 1991; 47:262–267. PMID: 1954984.

10. Chen ML, Yu WC, Lam CW, Au KM, Kong FY, Wo Cham AY. Diagnostic value of pleural fluid adenosine deaminase activity in tuberculous pleurisy. Clin Chim Acta. 2004; 341:101–107. PMID: 14967164.

11. Zaric B, Kuruc V, Milovancev A, Markovic M, Sarcev T, Canak V, et al. Differential diagnosis of tuberculosis and malignant pleural effusions: what is the role of adenosine deaminase? Lung. 2008; 186:233–240. PMID: 18357489.

12. Porcel JM, Aleman C, Bielsa S, Sarrapio J, Fernandez de Sevilla T, Esquerda A. A decision tree for differentiating tuberculous from malignant pleural effusions. Respir Med. 2008; 102:1159–1164. PMID: 18573650.

13. McKenna RW, Kyle RA, Kuehl WM, Grogan TM, Harris NL, Coupland RW. Swerdlow SH, Campo E, Harris NL, editors. Plasma cell neoplasms. WHO classification of tumours of haematopoietic and lymphoid tissues. 2008. 4th ed. Lyon, France: IARC;p. 200–213.

14. Jancelewicz Z, Takatsuki K, Sugai S, Pruzanski W. IgD multiple myeloma. Review of 133 cases. Arch Intern Med. 1975; 135:87–93. PMID: 1111472.

15. Blade J, Lust JA, Kyle RA. Immunoglobulin D multiple myeloma: presenting features, response to therapy, and survival in a series of 53 cases. J Clin Oncol. 1994; 12:2398–2404. PMID: 7964956.

16. Pruzanski W, Rother I. IgD plasma cell neoplasia: clinical manifestations and characteristic features. Can Med Assoc J. 1970; 102:1061–1065. PMID: 5000054.

17. Johnston JB, Weinerman B, Cooney T, Bowman DM, Pettigrew NM, Orr K. IgD kappa plasma cell dyscrasias: extraosseous manifestations including isolated leptomeningitis. Am J Clin Pathol. 1982; 77:60–65. PMID: 6798858.

18. Kamal MK, Williams E, Poskitt TR. IgD myeloma with malignant pleural effusion. South Med J. 1987; 80:657–658. PMID: 3576276.

19. Kim YM, Lee KK, Oh HS, Park SK, Won JH, Hong DS, et al. Myelomatous effusion with poor response to chemotherapy. J Korean Med Sci. 2000; 15:243–246. PMID: 10803706.

20. Natori K, Izumi H, Nagase D, Fujimoto Y, Ishihara S, Kato M, et al. IgD myeloma indicated by plasma cells in the peripheral blood and massive pleural effusion. Ann Hematol. 2008; 87:587–589. PMID: 18265984.

21. Yokoyama T, Tanaka A, Kato S, Aizawa H. Multiple myeloma presenting initially with pleural effusion and a unique paraspinal tumor in the thorax. Intern Med. 2008; 47:1917–1920. PMID: 18981637.

22. Chang H, Chou WC, Lee SY, Huang JY, Hung YH. Myelomatous pleural effusion in a patient with plasmablastic myeloma: a case report. Diagn Cytopathol. 2009; 37:205–207. PMID: 19170175.

23. Cristalli G, Costanzi S, Lambertucci C, Lupidi G, Vittori S, Volpini R, et al. Adenosine deaminase: functional implications and different classes of inhibitors. Med Res Rev. 2001; 21:105–128. PMID: 11223861.

24. Meier J, Coleman MS, Hutton JJ. Adenosine deaminase activity in peripheral blood cells of patients with haematological malignancies. Br J Cancer. 1976; 33:312–319. PMID: 1063590.

25. Ohata M, Masuda I, Nonaka K, Sugiura K. Combination assay of IAP and ADA in hematologic malignancies. Rinsho Byori. 1990; 38:703–710. PMID: 2381093.

26. Shaughnessy J Jr, Tian E, Sawyer J, McCoy J, Tricot G, Jacobson J, et al. Prognostic impact of cytogenetic and interphase fluorescence in situ hybridization-defined chromosome 13 deletion in multiple myeloma: early results of total therapy II. Br J Haematol. 2003; 120:44–52. PMID: 12492575.

27. Chiecchio L, Protheroe RKM, Ibrahim AH, Cheung KL, Rudduck C, Dagrada GP, et al. Deletion of chromosome 13 detected by conventional cytogenetics is a critical prognostic factor in myeloma. Leukemia. 2006; 20:1610–1617. PMID: 16826223.