Skip to main content

Advertisement

SpringerLink
Log in

HTLV-1-Associated Adult T Cell Leukemia Lymphoma Presenting as Granulomatous Pneumocystis Jiroveci Pneumonia (PJP) and Hypercalcemia

  • Case Reports/Clinical Vignettes
  • Published:
Journal of General Internal Medicine Aims and scope Submit manuscript

Background

Since the initial description of human T cell lymphotropic virus (HTLV-1), clusters of this infection have been detected globally. Unlike HIV infection, most patients infected with HTLV-1 remain asymptomatic throughout their lifetime.

Case Report

We report the case of a 39-year-old Afro-Caribbean man with HTLV-1 infection presenting as hypercalcemia and granulomatous pneumocystis jiroveci pneumonia.

Results

Interestingly, the hypercalcemia presented with normal parathyroid hormone–related protein and low 1,25 dihydroxyvitamin D levels, and the presence of pneumocystis jiroveci in the granulomas was diagnosed with transbronchial biopsy taken during bronchoscopy. HTLV-1-associated adult T cell leukemia lymphoma (ATLL) was diagnosed in this patient by bone marrow and lymph node biopsy.

Conclusion

Increased bone resorption, likely cytokine-mediated, is the most likely mechanism of hypercalcemia in this patient. This is believed to be the first description of this type of reaction to pneumocystis jiroveci in a HTLV-1-infected ATLL patient.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  1. Watanabe T. HTLV-1-associated diseases. Int J Hematol. 1997; 66(3):257–8.

    Article  PubMed  CAS  Google Scholar 

  2. Shimoyama M. Diagnostic criteria and classification of clinical subtypes of adult T-cell leukemia-lymphoma. Br J Haematol. 1991; 79(3):428–37.

    Article  PubMed  CAS  Google Scholar 

  3. Moseley JM, Danks JA, GrillV, Lister TA, Horton MA. Immunocytochemical demonstration of PTHrP protein in neoplastic tissue of HTLV-1 positive human adult T cell leukemia/lymphoma: implications for the mechanism of hypercalcemia. Br J Cancer. 1991; 64(4):745–8.

    PubMed  CAS  Google Scholar 

  4. Rhew DC, Gaultier CR, Daar ES, Zakowski PC, Said J. Infections in patients with chronic adult T cell leukemia/lymphoma: case report and review. Clin Infect Dis. 1995; 21(4):1014–6.

    PubMed  CAS  Google Scholar 

  5. Fetchick DA, Bertolini DR, Sarin PS, Weintraub ST, Mundy GR, Dunn JF. Production of 1,25-dihydroxivitamin D3 by human T cell lymphotropic virus-I-transformed lymphocytes. J Clin Invest. 1986; 78(2):592–6.

    Article  PubMed  CAS  Google Scholar 

  6. Seymour JF, Younes A, Cabanilla F. Lymphomatous presentation of CD4+/CD8+ HTLV-1-related adult T-cell leukemia/lymphoma in an Iranian woman. Leuk Lymphoma. 1994; 12(56):471–6.

    Article  PubMed  CAS  Google Scholar 

  7. Prager D, Rosenblatt JD, Ejima E. Hypercalcemia, parathyroid hormone-related protein expression and human T-cell leukemia virus infection. Leuk Lymphoma. 1994; 14(56):395–400.

    PubMed  CAS  Google Scholar 

  8. Edwards CM, Edwards SJ, Bhumbra RP, Chowdhury TA. Severe refractory hypercalcemia in HTLV-1 infection. J R Soc Med. 2003; 96(3):126–7.

    Article  PubMed  Google Scholar 

  9. Grindstaff P, Gruener G. The peripheral nervous system complications of HTLV-1 myelopathy (HAM/TSP) syndromes. Semin Neurol. 2005; 25(3):315–27.

    Article  PubMed  Google Scholar 

  10. Ikeda K, Okazaki R, Inoue D, Ohno H, Ogata E, Matsumoto T. Interleukin-2 increases production and secretion of parathyroid hormone-related peptide by human T-cell leukemia virus type I-infected cells: possible role in hypercalcemia associated with adult T cell leukemia. Endocrinology. 1993; 132(6):2551–6.

    Article  PubMed  CAS  Google Scholar 

  11. Ikeda K, Okazaki R, Inoue D, Ogata E, Matsumoto T. Transcription of the gene for parathyroid hormone-related peptide from the human is activated through a cAMP-dependent pathway by prostaglandin E1 in HTLV-I-infected T cells. J Biol Chem. 1993; 268(2):1174–9.

    PubMed  CAS  Google Scholar 

  12. Watanabe T, Yamaguchi K, Takatsuki K Osame M, Yoshida M. Constitutive expression of parathyroid hormone-related protein gene in human T cell leukemia virus type 1 (HTLV-1) carriers and adult T cell leukemia patients that can be trans-activated by HTLV-1 tax gene. J Exp Med. 1990; 172(3):759–65.

    Article  PubMed  CAS  Google Scholar 

  13. Fukumoto S, Matsumoto T, Watanabe T, Takahashi H, Miyoshi I, Ogata E. Secretion of parathyroid hormone-like activity from human T-cell lymphotropic virus type I-infected lymphocytes. Cancer Res. 1989; 49(14):3849–52.

    PubMed  CAS  Google Scholar 

  14. Ejima E, Rosenblatt JD, Massari M, et al. Cell-type-specific transactivation of the parathyroid hormone-related protein gene promoter by the human T-cell leukemia virus type I (HTLV-I) tax and HTLV-II tax proteins. Blood. 1993; 81(4):1017–24.

    PubMed  CAS  Google Scholar 

  15. Inoue D, Matsumoto T, Ogata E, Ikeda K. 22-Oxacalcitriol, a noncalcemic analogue of calcitriol, suppresses both cell proliferation and parathyroid hormone-related peptide gene expression in human T cell lymphotropic virus type I-infected T cells. J Biol Chem. 1993; 268(22):16730–6.

    PubMed  CAS  Google Scholar 

  16. Tomonaga M. Adult T-cell leukemia and opportunistic infections. Intern Med. 1999; 38(2):83–5.

    Article  PubMed  CAS  Google Scholar 

  17. Bondoc AY, White DA. Granulomatous pneumocystis carinii pneumonia in patients with malignancy. Thorax. 2002; 57(5):435–7.

    Article  PubMed  CAS  Google Scholar 

  18. Cupples JB, Blackie SP, Road JD. Granulomatous pneumocystis carinii pneumonia mimicking tuberculosis. Arch Pathol Lab Med. 1989; 113(11):1281–4.

    PubMed  CAS  Google Scholar 

  19. Oki J, Kami M, Kishi Y, et al. Pneumocystis carinii pneumonia with an atypical granulomatous response in a patient with chronic lymphocytic leukemia. Leuk Lymphoma 2001; 41(3–4):435–8.

    PubMed  CAS  Google Scholar 

  20. Saldana MJ, Mones JM. Cavitation and other atypical manifestations of pneumocystis carinii pneumonia. Semin Diagn Pathol. 1989; 6(3):273–86.

    PubMed  CAS  Google Scholar 

  21. Fornos PS, Jenkinson SG. Pneumocystis carinii pneumonia mimicking granulomatous lung disease. J La State Med Soc. 1992; 144(1):35–8.

    PubMed  CAS  Google Scholar 

  22. Ullmer E, Mayr M, Binet I, et al. Granulomatous pneumocystis carinii pneumonia in Wegener’s granulomatosis. Eur Respir J. 2000; 15(1):213–6.

    PubMed  CAS  Google Scholar 

  23. Cruickshank B. Pulmonary granulomatous pneumocystosis following renal transplantation. Am J Clin Pathol. 1975; 63(3):384–90.

    PubMed  CAS  Google Scholar 

  24. Chen WC, Chang SC, Wu TH, Yang WC, Tarng DC. Hypercalcemia in a renal transplant recipient suffering with pneumocystis carinii pneumonia. Am J Kidney Dis. 2002; 39(2):E8.

    Article  PubMed  Google Scholar 

  25. Mills AK, Wright SJ, Taylor KM, McCormick JG. Hypercalcemia caused by pneumocystis carinii pneumonia while in leukemic remission. Aust N Z J Med. 1999; 29(1):102–3.

    PubMed  CAS  Google Scholar 

  26. Kester KE, Byrd JC, Rearden TP, Zacher LL, Cragun WH, Hargis JB. Granulomatous pneumocystis carinii pneumonia in patients with low-grade lymphoid malignancies: a diagnostic dilemma. Clin Infect Dis. 1996; 22(6):1111–2.

    PubMed  CAS  Google Scholar 

  27. Roudier M, Lamaury I, Strobel M. Human T cell leukemia/lymphoma virus type 1 (HTLV-1) and pneumocystis carinii associated with T cell proliferation and haemophagocytic syndrome. Leukemia 1997; 11(3): 453–4.

    Article  PubMed  CAS  Google Scholar 

  28. Guise TA, Yin JJ, Taylor SD, et al. Evidence for a causal role of paradthyroid hormone-related protein in the pathogenesis of human breast cancer-mediated osteolysis. J Clin Invest 1996; 98(7): 1544–9.

    Article  PubMed  CAS  Google Scholar 

Download references

Potential Financial Conflicts of Interest

None disclosed.

Author information

Authors and Affiliations

  1. Department of Medicine, Mount Sinai Services, Queens Hospital Center, 82-68 164th Street, Jamaica, NY, 11432, USA

    Sabiha Shahnaz MD, David Reich MD, Diana Arévalo-Valencia MD, Slavka Kucinska MD, Joanna Tulczynska MD & Jean Fleischman MD

  2. Mount Sinai School of Medicine, New York, NY, USA

    Sabiha Shahnaz MD, David Reich MD, Diana Arévalo-Valencia MD, Slavka Kucinska MD, Joanna Tulczynska MD & Jean Fleischman MD

Authors
  1. Sabiha Shahnaz MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. David Reich MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Diana Arévalo-Valencia MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Slavka Kucinska MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Joanna Tulczynska MD
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Jean Fleischman MD
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to David Reich MD.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Shahnaz, S., Reich, D., Arévalo-Valencia, D. et al. HTLV-1-Associated Adult T Cell Leukemia Lymphoma Presenting as Granulomatous Pneumocystis Jiroveci Pneumonia (PJP) and Hypercalcemia. J GEN INTERN MED 22, 420–423 (2007). https://doi.org/10.1007/s11606-006-0047-6

Download citation

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s11606-006-0047-6

Key words

Search

Navigation