Atypical Pneumocystis jiroveci pneumonia with multiple nodular granulomas after rituximab for refractory nephrotic syndrome

Mai, Sabine; Ito, Shuichi; Ogura, Masao; Kamei, Koichi; Miyairi, Isao; Miyata, Ippei; Higuchi, Masataka; Matsuoka, Katsuyoshi

doi:10.1007/s00467-012-2286-6

Cited by 49 publications

(25 citation statements)

References 23 publications

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“…Since no randomized controlled trials in adults have been performed to date, severe adverse events might be underestimated by these reports. In contrast, a pediatric FSGS patient with RTX-associated lung injury [48] and a 3-year-old boy with atypical Pneumocystis jiroveci pneumonia following RTX administration [51] on the basis of steroid-resistant nephrotic syndrome have been reported. One has to keep in mind that reactivation of hepatitis B virus (HBV) infection was observed following RTX [52].…”

Section: Discussionmentioning

confidence: 99%

Rituximab Treatment for Relapsing Minimal Change Disease and Focal Segmental Glomerulosclerosis: A Systematic Review

et al. 2014

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Background: Minimal change disease (MCD) and focal segmental glomerulosclerosis (FSGS) remain a therapeutic challenge, since steroids and other immunosuppressive agents exhibit an unfavorable adverse event spectrum. The aim of this review was to systematically summarize and analyze data from preexisting studies reporting the outcome of rituximab (RTX) treatment in these patients. Methods: Study data on adult patients with either steroid-dependent or frequently relapsing MCD/FSGS were identified by a PubMed and Embase search. The number of relapses was calculated and the use of immunosuppressive co-medication prior to and after RTX treatment was quantified. Results: We identified14 studiesincluding 86 patients with frequently relapsing and steroid-dependent MCD or FSGS. Treatment with RTX reduced the number of relapses per year from 1.3 (0-9) relapses prior to treatment compared to 0 (0-2) after therapy (p < 0.001). Proteinuria decreased from 2.43 (0-15) g/day to 0 (0-4.89) g/day (p < 0.001), while serum albumin increased from 2.9 (1.2-4.6) at baseline to 4.0 (1.8-5.09) g/l after RTX (p = 0.001). The use of immunosuppression used at the time of RTX administration was also reduced after RTX therapy (p < 0.001). Baseline serum albumin was lower (p = 0.018), whereas the number of immunosuppressants prior to RTX was higher (p = 0.018) in patients with relapse after RTX. Conclusions: The published data suggest that RTX is effective in reducing the number of relapses and sparing immunosuppression in frequently relapsing and steroid-dependent nephrotic syndrome due to MCD and FSGS. These promising findings have to be confirmed in controlled and prospective studies.

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Section: Discussionmentioning

confidence: 99%

Rituximab Treatment for Relapsing Minimal Change Disease and Focal Segmental Glomerulosclerosis: A Systematic Review

et al. 2014

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“…Also, depletion, or a decrease, of peripheral B cells can lead to onset of bacterial or viral infections, for which caution is required, particularly in children. In Japan, onset of atypical Pneumocystis jiroveci pneumonia after rituximab therapy was reported in a patient with refractory steroid-resistant nephrotic syndrome [88]. Use of sulfamethoxazole/trimethoprim during the period of peripheral B-cell depletion for prophylaxis of pneumocystis infection has been investigated [81,85,86].…”

Section: Rituximabmentioning

confidence: 98%

Clinical practice guideline for pediatric idiopathic nephrotic syndrome 2013: medical therapy

et al. 2015

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“…In order to mount an appropriate defense against P. jirovecii , both T and B lymphocytes are crucial to coordinate an inflammatory response, produce antibodies and eventually clear the microorganism [1,2,3,9]. Any defect in either the function or count of these cells can lead to P. jirovecii pneumonia.…”

Section: Discussionmentioning

confidence: 99%

“…A review of the available literature points out that bronchoscopy with cytological analysis is commonly considered to be of very low diagnostic yield, if any, to detect P. jirovecii granulomas, whereas lung biopsy, either open or transbronchial, is widely regarded as the gold standard [1,3,4,7,8]. From the cases described by Hartel et al [7], the false-negative rate that we can calculate is at best as high as 35% in open lung biopsies.…”

Section: Introductionmentioning

confidence: 99%

“…A few articles document its occurrence during the treatment of hematological and solid malignancies, typically when anti-CD20 drugs (such as rituximab) and/or corticosteroids are used. These drugs lower the circulating lymphocyte count and interfere with their function, thus impeding the usual lymphocyte-dependent defense against P. jirovecii [1,2,3]. It is of note that rituximab and corticosteroids are also used for the treatment of various conditions such as pediatric nephrotic syndrome, transplantation, rheumatoid arthritis or Wegener's granulomatosis, and articles documenting either diffuse alveolar or granulomatous forms of P. jirovecii pneumonia arising in the course of the treatment of these conditions can be found in the literature [1,4,5,6].…”

Section: Introductionmentioning

confidence: 99%

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Granulomatous Reaction to <b><i>Pneumocystis jirovecii</i></b> Diagnosed in a Bronchoalveolar Lavage: A Case Report

Nobile

Valenti²,

Aubert³

et al. 2015

Acta Cytologica

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Background: Granulomatous reaction to Pneumocystis jirovecii is a rare but well-known pathological finding encountered in the setting of immunosuppression, HIV infection being the most common cause. It can also potentially complicate the treatment of hematological malignancies, typically when drugs lowering the count and function of lymphocytes are used. Lung biopsy is considered the gold standard for the diagnosis of granulomatous P. jirovecii pneumonia, whereas the diffuse alveolar form is usually detected cytologically in bronchoalveolar lavage (BAL). Case: A female patient pursuing R-CHOP chemotherapy for the treatment of multiple hematological malignancies developed a rapidly progressing dyspnea. Chest CT scans revealed a worsening of a known infiltrative lung disease thought to be secondary to her chemotherapy. Alterations compatible with a drug-induced interstitial lung disease and well-formed focally necrotizing granulomas were observed on an open lung biopsy, but no microorganism was identified with special stains. Eventually, a granulomatous reaction to P. jirovecii was found in a BAL and allowed appropriate treatment with rapid improvement of the dyspnea. Conclusion: Because granulomas are tissue-bound structures, they are rarely described in BAL. This article describes the first reported cytological diagnosis of a granulomatous reaction to P. jirovecii and the potential diagnostic interest of such a peculiar finding.

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Atypical Pneumocystis jiroveci pneumonia with multiple nodular granulomas after rituximab for refractory nephrotic syndrome

Abstract: The indication of rituximab for refractory nephrotic syndrome has increased recently. Therefore, recognition of the risk of atypical PCP is important. Our findings suggest that PCP prophylaxis should be considered following rituximab therapy.

Cited by 49 publications

References 23 publications

Rituximab Treatment for Relapsing Minimal Change Disease and Focal Segmental Glomerulosclerosis: A Systematic Review

Rituximab Treatment for Relapsing Minimal Change Disease and Focal Segmental Glomerulosclerosis: A Systematic Review

Clinical practice guideline for pediatric idiopathic nephrotic syndrome 2013: medical therapy

Granulomatous Reaction to <b><i>Pneumocystis jirovecii</i></b> Diagnosed in a Bronchoalveolar Lavage: A Case Report

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