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Hypercalcemia Heralding Pneumocystis jirovecii Pneumonia in an HIV-Seronegative Patient with Diffuse Cutaneous Systemic Sclerosis

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Abstract

Pneumocystis pneumonia (PCP) is a life-threatening fungal infection occurring in immunocompromised patients such as HIV-positive patients with low CD4 cell count or patients under heavy immunosuppressive therapy. We report the case of a 59-year-old male with severe diffuse cutaneous systemic sclerosis presenting with asthenia, dry cough and worsening shortness of breath for the last 15 days. Biological studies were remarkable for PTH-independent severe hypercalcemia with low 25-hydroxyvitamin D and a paradoxically elevated 1,25-dihydroxyvitamin D. Early bronchoalveolar lavage allowed for PCP diagnosis and targeted treatment. We discuss the underlying physiopathology and difficulties regarding prophylaxis and treatment.

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References

  1. Fréalle E, Valade S, Guigue N, Hamane S, Chabé M, Le Gal S, et al. Diffusion of Pneumocystis jirovecii in the surrounding air of patients with Pneumocystis colonization: frequency and putative risk factors. Med Mycol. 2017;55(5):568–72.

    PubMed  Google Scholar 

  2. Choukri F, Menotti J, Sarfati C, Lucet JC, Nevez G, Garin YJ, Derouin F, Totet A. Quantification and spread of Pneumocystis jirovecii in the surrounding air of patients with Pneumocystis pneumonia. Clin Infect Dis. 2010;51(3):259–65.

    Article  CAS  Google Scholar 

  3. Ahmed B, Jaspan JB. Case report: hypercalcemia in a patient with AIDS and Pneumocystis carinii pneumonia. Am J Med Sci. 1993;306(5):313–6.

    Article  CAS  Google Scholar 

  4. Mills AK, Wright SJ, Taylor KM, McCormack JG. Hypercalcaemia caused by Pneumocystis carinii pneumonia while in leukaemic remission. Aust N Z J Med. 1999;29(1):102–3.

    Article  CAS  Google Scholar 

  5. 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  Google Scholar 

  6. Hung YM. Pneumocystis carinii pneumonia with hypercalcemia and suppressed parathyroid hormone levels in a renal transplant patient. Transplantation. 2006;81(4):639.

    Article  Google Scholar 

  7. Aguirre AR, Balbo BE, Ianhez LE, da Costa MC, Andrade L. Hypercalcemia and suppressed PTH levels in a renal transplant patient infected with Pneumocystis carinii. Ren Fail. 2007;29(4):513–6.

    Article  CAS  Google Scholar 

  8. Hajji K, Dalle F, Harzallah A, Tanter Y, Rifle G, Mousson C. Vitamin D metabolite–mediated hypercalcemia with suppressed parathormone concentration in Pneumocystis jiroveci pneumonia after kidney transplantation. Transplant Proc. 2009;41(8):3320–2.

    Article  CAS  Google Scholar 

  9. Bency R, Roger SD, Elder GJ. Hypercalcaemia as a prodromal feature of indolent Pneumocystis jivorecii after renal transplantation. Nephrol Dial Transplant. 2011;26(5):1740–2.

    Article  CAS  Google Scholar 

  10. Chatzikyrkou C, Clajus C, Haubitz M, Hafer C. Hypercalcemia and pneumocystis pneumonia after kidney transplantation: report of an exceptional case and literature review. Transpl Infect Dis. 2011;13(5):496–500.

    Article  CAS  Google Scholar 

  11. Ramalho J, Bacelar Marques ID, Aguirre AR, Pierrotti LC, de Paula FJ, Nahas WC, David-Neto E. Pneumocystis jirovecii pneumonia with an atypical granulomatous response after kidney transplantation. Transpl Infect Dis. 2014;16(2):315–9.

    Article  CAS  Google Scholar 

  12. Dubrofsky L, Lipman ML, Nessim SJ. The case| hypercalcemia in a renal transplant recipient. Kidney Int. 2015;88(5):1207–8.

    Article  Google Scholar 

  13. Ling J, Anderson T, Warren S, Kirkland G, Jose M, Yu R, et al. Hypercalcaemia preceding diagnosis of Pneumocystis jirovecii pneumonia in renal transplant recipients. Clin Kidney J. 2017;10(6):845–51.

    Article  CAS  Google Scholar 

  14. El-Reshaid K, Al-Bader S. Hypercalcemic crisis as a prodromal feature of Pneumcystis jirovecii pneumonia. Saudi J Kidney Dis Transpl. 2018;29(4):993–6.

    Article  Google Scholar 

  15. Taylor LN, Aesif SW, Matson KM. A case of Pneumocystis pneumonia, with a granulomatous response and Vitamin D-mediated hypercalcemia, presenting 13 years after renal transplantation. Transpl Infect Dis. 2019;20:e13081.

    Article  Google Scholar 

  16. Meyer KC, Raghu G, Baughman RP, Brown KK, Costabel U, du Bois RM, Drent M, Haslam PL, Kim DS, Nagai S, Rottoli P, Saltini C, Selman M, Strange C, Wood B, American Thoracic Society Committee on BAL in Interstitial Lung Disease. An official American Thoracic Society clinical practice guideline: the clinical utility of bronchoalveolar lavage cellular analysis in interstitial lung disease. Am J Respir Crit Care Med. 2012;185(9):1004–14.

    Article  Google Scholar 

  17. Carmona EM, Limper AH. Update on the diagnosis and treatment of Pneumocystis pneumonia. Ther Adv Respir Dis. 2011;5(1):41–59.

    Article  CAS  Google Scholar 

  18. Roux A, Canet E, Valade S, et al. Pneumocystis jirovecii pneumonia in patients with or without AIDS. Fr Emerg Infect Dis. 2014;20:1490–7.

    Article  Google Scholar 

  19. Li M-C, Lee N-Y, Lee C-C, Lee H-C, Chang C-M, Ko W-C. Pneumocystis jiroveci pneumonia in immunocompromised patients: delayed diagnosis and poor outcomes in non-HIV-infected individuals. J Microbiol Immunol Infect. 2014;47:42–7.

    Article  Google Scholar 

  20. Kadakia J, Kiyabu M, Sharma OP, Boylen T. Granulomatous response to Pneumocystis carinii in patients infected with HIV. Sarcoidosis. 1993;10(1):44–9.

    CAS  PubMed  Google Scholar 

  21. Thomas CF, Limper AH. Pneumocystis pneumonia. N Engl J Med. 2004;350(24):2487–98.

    Article  CAS  Google Scholar 

  22. Travis WD, Pittaluga S, Lipschik GY, et al. Atypical pathologic manifestations of Pneumocystis carinii pneumonia in the acquired immune deficiency syndrome. Review of 123 lung biopsies from 76 patients with emphasis on cysts, vascular invasion, vasculitis, and granulomas. Am J Surg Pathol. 1990;14(7):615–25.

    Article  CAS  Google Scholar 

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

    Article  CAS  Google Scholar 

  24. Lauffer L, Kini JA, Costello P, Godleski J. Granulomatous Pneumocystis carinii pneumonia in a non-AIDS patient: an atypical presentation. J Thorac Imaging. 2004;19(3):196–9.

    Article  Google Scholar 

  25. Dusso AS, Kamimura S, Gallieni M, et al. Gamma-interferon-induced resistance to 1,25-(OH)2 D3 in human monocytes and macrophages: a mechanism for the hypercalcemia of various granulomatoses. J Clin Endocrinol Metab. 1997;82(7):2222–32.

    CAS  PubMed  Google Scholar 

  26. Lionakis MS, Samonis G, Kontoyiannis DP. Endocrine and metabolic manifestations of invasive fungal infections and systemic antifungal treatment. Mayo Clin Proc. 2008;83(9):1046–60.

    Article  CAS  Google Scholar 

  27. Blaine J, Chonchol M, Levi M. Renal control of calcium, phosphate, and magnesium homeostasis. Clin J Am Soc Nephrol. 2015;10(7):1257–72. https://doi.org/10.2215/CJN.09750913.

    Article  CAS  Google Scholar 

  28. Baylis PH, Milles JJ, Wilkinson R, Heath DA. Vasopressin function in hypercalcaemia. Clin Endocrinol. 1981;15:343–51.

    Article  CAS  Google Scholar 

  29. Garofeanu CG, Weir M, Rosas-Arellano MP, et al. Causes of reversible nephrogenic diabetes insipidus: a systematic review. Am J Kidney Dis. 2005;45:626–37.

    Article  Google Scholar 

  30. Ward MM, Donald F. Pneumocystis carinii pneumonia in patients with connective tissue diseases: the role of hospital experience in diagnosis and mortality. Arthritis Rheum. 1999;42(4):780–9.

    Article  CAS  Google Scholar 

  31. Stern A, Green H, Paul M, Vidal L, Leibovici L. Prophylaxis for Pneumocystis pneumonia (PCP) in non-HIV immunocompromised patients. Cochrane Database Syst Rev. 2014;10:CD005590.

    Google Scholar 

  32. Messiaen PE, Cuyx S, Dejagere T, van der Hilst JC. The role of CD4 cell count as discriminatory measure to guide chemoprophylaxis against Pneumocystis jirovecii pneumonia in human immunodeficiency virus-negative immunocompromised patients: a systematic review. Transpl Infect Dis. 2017;19(2):e12651.

    Article  Google Scholar 

  33. Mecoli CA, Saylor D, Gelber AC, Christopher-Stine L. Pneumocystis jiroveci pneumonia in rheumatic disease: a 20-year single-centre experience. Clin Exp Rheumatol. 2017;35(4):671–3.

    PubMed  Google Scholar 

  34. Alexandre K, Ingen-Housz-Oro S, Versini M, Sailler L, Benhamou Y. Pneumocystis jirovecii pneumonia in patients treated with rituximab for systemic diseases: report of 11 cases and review of the literature. Eur J Intern Med. 2018;50:e23–4.

    Article  CAS  Google Scholar 

  35. Elsegeiny W, Eddens T, Chen K, Kolls JK. Anti-CD20 antibody therapy and susceptibility to Pneumocystis pneumonia. Infect Immun. 2015;83(5):2043–52.

    Article  CAS  Google Scholar 

  36. Green H, Paul M, Vidal L, Leibovici L. Prophylaxis of Pneumocystis pneumonia in immunocompromised non-HIV-infected patients: systematic review and meta-analysis of randomized controlled trials. Mayo Clin Proc. 2007;82:1052–9.

    Article  CAS  Google Scholar 

  37. Kosaka M, Ushiki A, Ikuyama Y, Hirai K, Matsuo A, Hachiya T, Hanaoka M. A four-center retrospective study of the efficacy and toxicity of low-dose trimethoprim-sulfamethoxazole for the treatment of Pneumocystis pneumonia in patients without HIV infection. Antimicrob Agents Chemother. 2017;61(12):e01173-17.

    Article  Google Scholar 

  38. Shibata T, Tonooka K, Tsuchida K, Mitomi H, Shibata T, Katsuyama N. Retrospective investigation of side effects and prognoses of moderate-dose trimethoprim-sulfamethoxazole treatment for pneumocystis pneumonia that developed in patients with autoimmune diseases. Nihon Rinsho Meneki Gakkai Kaishi. 2016;39(3):213–8.

    Article  CAS  Google Scholar 

  39. Bouchara JP, Chaturvedi V. The curious case of “case report” of infections caused by human and animal fungal pathogens: an educational tool, an online archive, or a format in need of retooling. Mycopathologia. 2018;183(6):879–91.

    Article  Google Scholar 

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Correspondence to Quentin Binet.

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The authors hereby certify that there is no conflict of interest regarding the publication of this paper. It has been written taking into account the checklist implemented by Mycopathologia to ensure consistency and quality across case reports [39].

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Binet, Q., Mairesse, J., Vanthuyne, M. et al. Hypercalcemia Heralding Pneumocystis jirovecii Pneumonia in an HIV-Seronegative Patient with Diffuse Cutaneous Systemic Sclerosis. Mycopathologia 184, 787–793 (2019). https://doi.org/10.1007/s11046-019-00397-w

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