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

Shahnaz, Sabiha; Reich, David; Arévalo-Valencia, Diana; Kucinska, Slavka; Tulczynska, Joanna M; Fleischman, Jean K.

doi:10.1007/s11606-006-0047-6

Cited by 17 publications

(13 citation statements)

References 27 publications

(33 reference statements)

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“…HTLV-1-related opportunistic infections are widely recognized; indeed, CMV gastritis, gastroenterocolitis [ 13 , 14 ], PCP [ 15 ], and disseminated herpes zoster [ 16 ] have all been reported. There have also been reports of strongyloidiasis [ 17 ], pulmonary histoplasmosis [ 18 ], and cryptococcal meningitis [ 19 ].…”

Section: Discussionmentioning

confidence: 99%

Opportunistic Infections in Patients with HTLV-1 Infection

Tanaka¹,

Sekioka²,

Usui³

et al. 2015

Case Reports in Hematology

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As an acquired immunodeficiency, human immunodeficiency virus (HIV) infection is primarily responsible for opportunistic infections in infected patients. However, opportunistic infections also occur in individuals with human T cell lymphotrophic virus type 1 (HTLV-1) infection. Here, we report opportunistic infections in two Japanese HTLV-1-seropositive patients. The first patient was a 67-year-old male, who had cytomegalovirus infection associated with esophagogastritis and terminal ileitis. The patient was HTLV-1-positive and was diagnosed with smoldering adult T cell leukemia (ATL). High levels of serum soluble IL-2 receptor (sIL-2R; 4,304 U/mL) and an increased percentage of CD4+CD25+ T cells (75.5%) in peripheral blood were also detected. The second patient was a 78-year-old female, a known asymptomatic HTLV-1 carrier, who presented with persistent herpes zoster, followed by Pneumocystis jirovecii pneumonia. Disease progression of smoldering ATL along opportunistic infections was observed with very high levels of serum sIL-2R (14,058 U/mL) and an increased percentage of CD4+CD25+ T cells (87.2%) in peripheral blood. In patients with suspected opportunistic infections, both HTLV-1 and HIV should be considered. In HTLV-1-positive patients, an increase in the CD4+CD25+ T cell subset may have its value as a prognostic marker.

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Section: Discussionmentioning

confidence: 99%

Opportunistic Infections in Patients with HTLV-1 Infection

Tanaka¹,

Sekioka²,

Usui³

et al. 2015

Case Reports in Hematology

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“…Likely, HTLV-1 infection will cause chronic immunosuppresion and activate B-cell proliferation, favoring the development of EBV infection, which in turn will prepare the host for the development of B-cell lymphoma portending a worse prognosis. It seems intuitive that a patient with a combination of severe immunological impairment due to EBV and HTLV-1 infections, immunosenescence and chemotherapy would develop life-threatening opportunistic infections, although the occurrence of PJP in HTLV-1 carriers has seldom been reported in the medical literature [ 31 , 32 ]. Further research is needed to better understand the interaction between EBV and HTLV-1 in lymphomagenesis.…”

Section: Resultsmentioning

confidence: 99%

EBV-positive diffuse large B-cell lymphoma in a human T-lymphotropic virus type 1 carrier

Beltrán

Salas

Quiñones

et al. 2009

Infect Agents Cancer

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The development of B-cell lymphomas has been seldom described in HTLV-1 carriers. We present the case of an elderly Peruvian HTLV-1 carrier who was diagnosed with EBV-positive diffuse large B-cell lymphoma. Despite an initial good response to therapy, patient died during treatment due to fatal Pneumocystis jirovecci pneumonia. EBV infection is characterized by B-cell lymphotropism and selective immunodeficiency. HTLV-1, on the other hand, induces T-cell dysfunction and B-cell proliferation. Finally, immunosenescence is characterized by T-cell dysregulation, decreased apoptosis and cytokine upregulation. In this elderly patient, the combination of EBV and HTLV-1 coinfection and immunosenescence may have played a role in the development of this aggressive diffuse large B-cell lymphoma. Furthermore, the immunodeficiency caused by the viral infections and chemotherapy may have played a role in developing life-threatening infectious complications. FindingsThe Epstein Barr virus (EBV) was the first described oncovirus, which has been associated with the development of a variety of lymphoproliferative disorders, such as Burkitt . EBV infection occurs early in childhood, and approximately 90 to 95% of adults worldwide are EBV-seropositive. EBV expression has also been reported in patients with diffuse large B-cell lymphoma (DLBCL) [6]. DLBCL is the most common variant of nonHodgkin lymphoma in the United States (US) and accounts for approximately 25-30% of the cases [7]. In Peru, DLBCL accounts for up to 45% of all lymphomas and, akin to Asian countries, there is high incidence of Tcell lymphomas and low incidence of follicular lymphomas [8]. On the other hand, the human T-lymphotropic virus type 1 (HTLV-1) is a retrovirus and is the pathogenic agent of adult T-cell lymphoma/leukemia (ATLL) and other diseases [9]. HTLV-1 is endemic in Japan, the Melanesian Islands, the Caribbean, South America, the Middle East and parts of Africa. The prevalence of HTLV-1 in Europe and the US is lower than 1%. In Peru, it is estimated that up to 3% of the healthy adult population carry HTLV-1 [10]. The interaction of these two oncoviruses, EBV and HTLV-1, has seldom been reported in the medical literature.

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“…The other populations described in the literature presenting PJP-induced hypercalcaemia are paediatric, AIDS and leukaemic patients 34–37. Interestingly, a common characteristic of these population is the immaturity or the dysfunction of lymphocyte T response, as in transplant recipients 28…”

Section: Discussionmentioning

confidence: 99%

Pneumocystis jiroveciipneumonitis: cause of acute hypercalcaemia in chronic haemodialysis patient

et al. 2020

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A 76-year-old renal transplant patient due to autosomal dominant polycystic kidney disease who resumed chronic haemodialysis was admitted to our hospital for confusion and lassitude. He was afebrile and physical examination revealed diffuse bilateral rales with decreased respiratory sounds in lower right lung. Laboratory data showed hypercalcaemia (total calcium 3.92 mmol/L (normal range 2.2–2.6 mmol/L), ionised calcium 1.87 mmol/L (1.15–1.35 mmol/L)), low intact parathyroid hormone (iPTH) 15 ng/L, (15–65 ng/L) and high 1,25(OH)2D3 128.9 pg/mL, (15.2–90.1 pg/mL). Chest CT-scan revealed bilateral apical lung lesions after 15 days of antibiotics. Bronchoalveolar sample was PCR positive for Pneumocystis jirovecii. He was treated with an extra session of haemodialysis with 1.25 mmol/L dialysate calcium concentration, oral trimethoprim-sulfamethoxazole was started and oral corticosteroid dose increased to 1 mg/kg for 1 week. Hypercalcaemia decreased progressively after initiation of these treatments. We concluded a case of hypercalcaemia secondary to P. jirovecii infection.

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

Cited by 17 publications

References 27 publications

Opportunistic Infections in Patients with HTLV-1 Infection

Opportunistic Infections in Patients with HTLV-1 Infection

EBV-positive diffuse large B-cell lymphoma in a human T-lymphotropic virus type 1 carrier

Pneumocystis jiroveciipneumonitis: cause of acute hypercalcaemia in chronic haemodialysis patient

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