PMID- 33312857
OWN - NLM
STAT- PubMed-not-MEDLINE
LR  - 20201216
IS  - 2213-0071 (Print)
IS  - 2213-0071 (Electronic)
IS  - 2213-0071 (Linking)
VI  - 31
DP  - 2020
TI  - Effect of everolimus on multifocal micronodular pneumocyte hyperplasia in 
      tuberous sclerosis complex.
PG  - 101310
LID - 10.1016/j.rmcr.2020.101310 [doi]
LID - 101310
AB  - Multifocal micronodular pneumocyte hyperplasia (MMPH) is a benign proliferation 
      of alveolar type II cells presenting as multiple pulmonary nodules at chest 
      imaging, which is frequently seen in patients with tuberous sclerosis complex 
      (TSC). We report a case of a woman with TSC and MMPH who received everolimus, a 
      mechanistic target of rapamycin (mTOR) inhibitor, for the treatment of a 
      subependymal giant cell astrocytoma (SEGA). After 3 months of therapy, a 
      remarkable decrease in density of all pulmonary MMPH lesions was observed, 
      without any change in size. This shows that everolimus is active on MMPH 
      similarly to its effects on SEGA, renal angiomyolipomas, and pulmonary 
      lymphangioleiomyomatosis in TSC, and suggests that the dysregulated activation of 
      mTOR which characterizes TSC also plays a role in the pathogenesis of MMPH.
CI  - (c) 2020 The Authors.
FAU - Daccord, Cecile
AU  - Daccord C
AD  - Department of Respiratory Medicine, Lausanne University Hospital (CHUV) and 
      University of Lausanne, Switzerland.
FAU - Nicolas, Aymeric
AU  - Nicolas A
AD  - Department of Radiodiagnostic and Interventional Radiology, Lausanne University 
      Hospital (CHUV) and University of Lausanne, Switzerland.
FAU - Demicheli, Rita
AU  - Demicheli R
AD  - Department of Oncology, Lausanne University Hospital (CHUV) and University of 
      Lausanne, Switzerland.
FAU - Chehade, Hassib
AU  - Chehade H
AD  - Department Woman-Mother-Child, Division of Paediatrics, Paediatric Nephrology 
      Unit, Lausanne University Hospital (CHUV) and University of Lausanne, 
      Switzerland.
FAU - Hottinger, Andreas F
AU  - Hottinger AF
AD  - Department of Oncology, Lausanne University Hospital (CHUV) and University of 
      Lausanne, Switzerland.
AD  - Department of Clinical Neurosciences, Lausanne University Hospital (CHUV) and 
      University of Lausanne, Switzerland.
FAU - Beigelman, Catherine
AU  - Beigelman C
AD  - Department of Radiodiagnostic and Interventional Radiology, Lausanne University 
      Hospital (CHUV) and University of Lausanne, Switzerland.
FAU - Lazor, Romain
AU  - Lazor R
AD  - Department of Respiratory Medicine, Lausanne University Hospital (CHUV) and 
      University of Lausanne, Switzerland.
LA  - eng
PT  - Case Reports
DEP - 20201125
PL  - England
TA  - Respir Med Case Rep
JT  - Respiratory medicine case reports
JID - 101604463
PMC - PMC7720070
OTO - NOTNLM
OT  - Alveolar epithelial cells
OT  - Everolimus
OT  - Lung
OT  - Mechanistic target of rapamycin
OT  - Multifocal micronodular pneumocyte hyperplasia
OT  - Tuberous sclerosis
COIS- The authors have no conflicts of interest to declare related to this work. AN, RD 
      and HC have nothing to disclose. CD reports non-financial support from 
      Boehringer-Ingelheim and Roche, outside the submitted work. AFH reports grants 
      from Novocure, personal fees from Karyopharm and BMS, outside the submitted work. 
      CB reports personal fees from Boehringer-Ingelheim, AstraZeneca, and Gilead 
      Sciences, outside the submitted work. RL reports personal fees and non-financial 
      support from Boehringer-Ingelheim, non-financial support from Roche and Vifor, 
      outside the submitted work.
EDAT- 2020/12/15 06:00
MHDA- 2020/12/15 06:01
PMCR- 2020/11/25
CRDT- 2020/12/14 10:58
PHST- 2020/07/02 00:00 [received]
PHST- 2020/11/22 00:00 [accepted]
PHST- 2020/12/14 10:58 [entrez]
PHST- 2020/12/15 06:00 [pubmed]
PHST- 2020/12/15 06:01 [medline]
PHST- 2020/11/25 00:00 [pmc-release]
AID - S2213-0071(20)30524-4 [pii]
AID - 101310 [pii]
AID - 10.1016/j.rmcr.2020.101310 [doi]
PST - epublish
SO  - Respir Med Case Rep. 2020 Nov 25;31:101310. doi: 10.1016/j.rmcr.2020.101310. 
      eCollection 2020.