Indication for a Pneumocystis Prophylaxis Therapy in Patients with Vascular Anomalies Treated with PIK3/AKT/mTOR Pathway Inhibitors: Experts' Opinion and Systematic Review from the Literature.


Journal

Dermatology (Basel, Switzerland)
ISSN: 1421-9832
Titre abrégé: Dermatology
Pays: Switzerland
ID NLM: 9203244

Informations de publication

Date de publication:
2023
Historique:
received: 23 11 2022
accepted: 14 08 2023
medline: 7 12 2023
pubmed: 5 10 2023
entrez: 4 10 2023
Statut: ppublish

Résumé

Vascular anomalies (VAs) are increasingly being treated with PI3K/AKT/mTOR pathway inhibitors. These drugs have immunosuppressive properties and thus theoretically overexpose patients to opportunistic infections, especially Pneumocystis jirovecii pneumonia (PJP). PJP prophylaxis use lacks consensus. We aimed to investigate the prevalence of PJP in patients receiving mTOR/PI3K/AKT inhibitors for VAs and determine any indication for pneumocystis prophylaxis in this population. The study was conducted in 2 parts: (1) we sent a survey to a panel of international experts of VAs asking about their use of pneumocystis prophylaxis drugs and (2) we performed a systematic review of the literature of all published cases of patients receiving these drugs for VA to estimate the prevalence of PJP in this population. Answers from 68 experts were analyzed: 21 (30.9%) answered they always add PJP prophylaxis when prescribing mTOR inhibitors, 20 (29.4%) case-by-case, and 27 (39.7%) never. For the systematic review, among 3,053 reports screened, 217 were included involving 1,189 patients (1,143 received sirolimus, 38 everolimus, 4 alpelisib, 4 miransertib). Among the 1,189 cases, 2 (0.2%) PJP were reported: one under sirolimus and one under everolimus. Thus, the prevalence of PJP was estimated at 0.88 cases/1,000 patients under sirolimus (95% CI: -0.84 to 2.59) and 26.31 cases/1,000 under everolimus (95% CI: -24.58 to 77.18). Patients with PJP never received prophylaxis drugs. We found no PJP cases under alpelisib and miransertib. PJP prophylaxis was given in 218 (18.3%) cases, more frequently for children (91.3 vs. 77.2% in the non-prophylaxis group, p = 0.012), mostly trimethoprim-sulfamethoxazole (186 patients, 85.3%). Our study shows that even if PJP is a rare event, it may occur in patients with VAs treated with an mTOR inhibitor. Although our results cannot allow for revising guidelines, prophylaxis with TMP-SMX might be appropriate for a subgroup of patients with risk factors for PJP.

Sections du résumé

BACKGROUND BACKGROUND
Vascular anomalies (VAs) are increasingly being treated with PI3K/AKT/mTOR pathway inhibitors. These drugs have immunosuppressive properties and thus theoretically overexpose patients to opportunistic infections, especially Pneumocystis jirovecii pneumonia (PJP). PJP prophylaxis use lacks consensus. We aimed to investigate the prevalence of PJP in patients receiving mTOR/PI3K/AKT inhibitors for VAs and determine any indication for pneumocystis prophylaxis in this population.
METHODS METHODS
The study was conducted in 2 parts: (1) we sent a survey to a panel of international experts of VAs asking about their use of pneumocystis prophylaxis drugs and (2) we performed a systematic review of the literature of all published cases of patients receiving these drugs for VA to estimate the prevalence of PJP in this population.
RESULTS RESULTS
Answers from 68 experts were analyzed: 21 (30.9%) answered they always add PJP prophylaxis when prescribing mTOR inhibitors, 20 (29.4%) case-by-case, and 27 (39.7%) never. For the systematic review, among 3,053 reports screened, 217 were included involving 1,189 patients (1,143 received sirolimus, 38 everolimus, 4 alpelisib, 4 miransertib). Among the 1,189 cases, 2 (0.2%) PJP were reported: one under sirolimus and one under everolimus. Thus, the prevalence of PJP was estimated at 0.88 cases/1,000 patients under sirolimus (95% CI: -0.84 to 2.59) and 26.31 cases/1,000 under everolimus (95% CI: -24.58 to 77.18). Patients with PJP never received prophylaxis drugs. We found no PJP cases under alpelisib and miransertib. PJP prophylaxis was given in 218 (18.3%) cases, more frequently for children (91.3 vs. 77.2% in the non-prophylaxis group, p = 0.012), mostly trimethoprim-sulfamethoxazole (186 patients, 85.3%).
CONCLUSION CONCLUSIONS
Our study shows that even if PJP is a rare event, it may occur in patients with VAs treated with an mTOR inhibitor. Although our results cannot allow for revising guidelines, prophylaxis with TMP-SMX might be appropriate for a subgroup of patients with risk factors for PJP.

Identifiants

pubmed: 37793356
pii: 000533675
doi: 10.1159/000533675
doi:

Substances chimiques

Alpelisib 08W5N2C97Q
Miransertib T1DQI1B52Y
MTOR Inhibitors 0
Proto-Oncogene Proteins c-akt EC 2.7.11.1
Everolimus 9HW64Q8G6G
Phosphatidylinositol 3-Kinases EC 2.7.1.-
Trimethoprim, Sulfamethoxazole Drug Combination 8064-90-2
TOR Serine-Threonine Kinases EC 2.7.11.1

Types de publication

Systematic Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

942-951

Informations de copyright

© 2023 S. Karger AG, Basel.

Auteurs

Maxime Navarro (M)

CHRU Tours, Department of Dermatology, Unit of Pediatric dermatology, Tours, France.
Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC-Tours), Tours, France.

Aude Allemang-Trivalle (A)

University of Tours, University of Nantes, INSERM 1246-SPHERE, Tours, France.

Sophie Leducq (S)

CHRU Tours, Department of Dermatology, Unit of Pediatric dermatology, Tours, France.
Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC-Tours), Tours, France.
University of Tours, University of Nantes, INSERM 1246-SPHERE, Tours, France.

Annie-Pierre Jonville-Bera (AP)

University of Tours, University of Nantes, INSERM 1246-SPHERE, Tours, France.
CHRU Tours, Department of Clinical Pharmacology, Regional Pharmacovigilance Center, Tours, France.

Anaïs Maurier (A)

CHRU Tours, Department of Clinical Pharmacology, Regional Pharmacovigilance Center, Tours, France.

Tarik Zejli (T)

CHRU Tours, Clinical Investigation Center of Tours, INSERM 1415, Bretonneau Hospital, Tours, France.

Afi-Emiliène Edée (AE)

Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC-Tours), Tours, France.

Emilie Harchaoui (E)

CHRU Tours, Department of Dermatology, Unit of Pediatric dermatology, Tours, France.
University of Tours, University of Nantes, INSERM 1246-SPHERE, Tours, France.

Bruno Giraudeau (B)

University of Tours, University of Nantes, INSERM 1246-SPHERE, Tours, France.
CHRU Tours, Clinical Investigation Center of Tours, INSERM 1415, Bretonneau Hospital, Tours, France.

Annabel Maruani (A)

CHRU Tours, Department of Dermatology, Unit of Pediatric dermatology, Tours, France.
Reference Center for Genodermatoses and Rare Skin Diseases (MAGEC-Tours), Tours, France.
University of Tours, University of Nantes, INSERM 1246-SPHERE, Tours, France.

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Classifications MeSH