How are you taking part in this consultation?

You will not be able to change how you comment later.

You must be signed in to answer questions

    The content on this page is not current guidance and is only for the purposes of the consultation process.

    Methods

    NICE identified studies and reviews relevant to phrenic nerve pacing for congenital central hypoventilation syndrome from the medical literature. The following databases were searched between the date they started to 11 October 2023: MEDLINE, PREMEDLINE, EMBASE, Cochrane Library and other databases. Trial registries and the internet were also searched (see the literature search strategy). Relevant published studies identified during consultation or resolution that are published after this date may also be considered for inclusion.

    The following inclusion criteria were applied to the abstracts identified by the literature search.

    • Publication type: clinical studies were included with emphasis on identifying good quality studies. Reviews, editorials, and laboratory or animal studies, were also excluded and so were conference abstracts, because of the difficulty of appraising study methodology, unless they reported specific adverse events that not available in the published literature.

    • Patients with congenital central hypoventilation syndrome.

    • Intervention or test: phrenic nerve pacing.

    • Outcome: articles were retrieved if the abstract contained information relevant to the safety, efficacy, or both.

    If selection criteria could not be determined from the abstracts the full paper was retrieved.

    Potentially relevant studies not included in the main evidence summary are listed in the section on other relevant studies.

    Find out more about how NICE selects the evidence for the committee.

    Table 4 literature search strategy

    Databases

    Date searched

    Version/files

    MEDLINE ALL (Ovid)

    11/10/2023

    1946 to October 10, 2023

    EMBASE (Ovid)

    11/10/2023

    1974 to 2023 October 10

    Cochrane Database of Systematic Reviews – CDSR (Cochrane Library)

    11/10/2023

    Issue 10 of 12, October 2023

    Cochrane Central Database of Controlled Trials – CENTRAL (Cochrane Library)

    11/10/2023

    Issue 10 of 12, October 2023

    International HTA database (INAHTA)

    11/10/2023

    -

    Trial sources searched:

    • Clinicaltrials.gov

    • ISRCTN

    • WHO International Clinical Trials Registry

    Websites searched:

    • National Institute for Health and Care Excellence (NICE)

    • NHS England

    • Food and Drug Administration (FDA) - MAUDE database

    • Australian Safety and Efficacy Register of New Interventional Procedures – Surgical (ASERNIP – S)

    • Australia and New Zealand Horizon Scanning Network (ANZHSN)

    • General internet search

    The following search strategy was used to identify papers in MEDLINE. A similar strategy was used to identify papers in other databases.

    MEDLINE search strategy

    1 Phrenic Nerve/

    2 Diaphragm/

    3 1 or 2

    4 (pacing or stimulat*).tw.

    5 3 and 4

    6 ((phrenic nerve* or diaphragm*) adj4 (pacing or stimulat*)).tw.

    7 5 or 6

    8 ((Congenital adj2 central adj2 hypoventilation adj2 syndrome*) or CCHS).tw.

    9 (Primary adj2 central adj2 hypoventilation adj2 syndrome*).tw.

    10 (Central adj4 hypoventilation).tw.

    11 "Ondine's curse".tw.

    12 or/8-11

    13 Spinal Cord Injuries/

    14 ((Spinal adj4 cord adj4 (injur* or trauma* or contusion* or lacerat*)) or SCI).tw.

    15 Respiration, Artificial/

    16 (((artificial or mechanical) adj4 ventilat*) or (Ventilat* adj4 dependen*)).tw.

    17 Respiratory Insufficiency/

    18 (Respiratory adj4 (failure* or artificial or Insufficien* or depression)).tw.

    19 or/13-18

    20 12 or 19

    21 7 and 20

    22 (Avery adj4 Diaphragm adj4 (Pace* or pacing)).tw.

    23 ((Atrostim or atrotech) adj4 (Phrenic adj4 Nerve)).tw.

    24 21 or 22 or 23

    25 Animals/ not Humans/

    26 24 not 25

    27 limit 26 to english language