PMID- 34703757
OWN - NLM
STAT- PubMed-not-MEDLINE
LR  - 20211028
IS  - 2213-0071 (Print)
IS  - 2213-0071 (Electronic)
IS  - 2213-0071 (Linking)
VI  - 34
DP  - 2021
TI  - Resolution of multifocal micronodular pneumocyte hyperplasia with everolimus in a 
      patient with tuberous sclerosis complex.
PG  - 101526
LID - 10.1016/j.rmcr.2021.101526 [doi]
LID - 101526
AB  - A woman with a diagnosis of tuberous sclerosis complex (TSC) presented with TSC2 
      gene mutation and various manifestations, including epilepsy, renal 
      angiomyolipomas (AML), and pathologically confirmed multifocal micronodular 
      pneumocyte hyperplasia (MMPH). With oral administration of everolimus, a 
      mammalian target of rapamycin (mTOR) inhibitor, MMPH and AML were markedly 
      reduced. Further, after starting treatment with everolimus, serum levels of 
      surfactant protein (SP)-A and SP-D, which reflect type II pneumocyte hyperplasia, 
      decreased to the normal range. At the time of writing of this manuscript, 6 years 
      after starting everolimus, MMPH lesions did not relapse and SP-A/D remained the 
      low levels. This is the first case of everolimus efficacy shown for 
      histologically confirmed MMPH in genetically determined TSC patient, with time 
      course of serum SP-A and SP-D.
CI  - (c) 2021 Published by Elsevier Ltd.
FAU - Shoji, Tetsuaki
AU  - Shoji T
AD  - Department of Respiratory Medicine, Faculty of Medicine and Graduate School of 
      Medicine, Hokkaido University, Sapporo, Hokkaido, Japan.
FAU - Niida, Yo
AU  - Niida Y
AD  - Center for Clinical Genomics, Kanazawa Medical University Hospital, Ishikawa, 
      Japan.
FAU - Osawa, Takahiro
AU  - Osawa T
AD  - Department of Urology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Hokkaido, Japan.
FAU - Matsumoto, Ryuji
AU  - Matsumoto R
AD  - Department of Urology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Hokkaido, Japan.
FAU - Sakurai, Kotaro
AU  - Sakurai K
AD  - Department of Psychiatry, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Hokkaido, Japan.
FAU - Suzuki, Masaru
AU  - Suzuki M
AD  - Department of Respiratory Medicine, Faculty of Medicine and Graduate School of 
      Medicine, Hokkaido University, Sapporo, Hokkaido, Japan.
FAU - Matsuno, Yoshihiro
AU  - Matsuno Y
AD  - Department of Surgical Pathology, Hokkaido University Hospital, Sapporo, 
      Hokkaido, Japan.
FAU - Konno, Satoshi
AU  - Konno S
AD  - Department of Respiratory Medicine, Faculty of Medicine and Graduate School of 
      Medicine, Hokkaido University, Sapporo, Hokkaido, Japan.
LA  - eng
PT  - Case Reports
DEP - 20211006
PL  - England
TA  - Respir Med Case Rep
JT  - Respiratory medicine case reports
JID - 101604463
PMC - PMC8524238
OTO - NOTNLM
OT  - Everolimus
OT  - Multifocal micronodular pneumocyte hyperplasia
OT  - Surfactant Protein-A (SP-A)
OT  - Surfactant protein-B (SP-B)
OT  - Tuberous sclerosis complex
COIS- All authors inform that there is no conflict of interest.
EDAT- 2021/10/28 06:00
MHDA- 2021/10/28 06:01
PMCR- 2021/10/06
CRDT- 2021/10/27 07:09
PHST- 2021/08/03 00:00 [received]
PHST- 2021/09/20 00:00 [revised]
PHST- 2021/10/04 00:00 [accepted]
PHST- 2021/10/27 07:09 [entrez]
PHST- 2021/10/28 06:00 [pubmed]
PHST- 2021/10/28 06:01 [medline]
PHST- 2021/10/06 00:00 [pmc-release]
AID - S2213-0071(21)00188-X [pii]
AID - 101526 [pii]
AID - 10.1016/j.rmcr.2021.101526 [doi]
PST - epublish
SO  - Respir Med Case Rep. 2021 Oct 6;34:101526. doi: 10.1016/j.rmcr.2021.101526. 
      eCollection 2021.