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Prevention of morbidity in sickle cell disease-qualitative outcomes, pain and quality of life in a randomised cross-over pilot trial of overnight supplementary oxygen and auto-adjusting continuous positive airways pressure (POMS2a): study protocol for a randomised controlled trial

Prevention of morbidity in sickle cell disease-qualitative outcomes, pain and quality of life in a randomised cross-over pilot trial of overnight supplementary oxygen and auto-adjusting continuous positive airways pressure (POMS2a): study protocol for a randomised controlled trial
Prevention of morbidity in sickle cell disease-qualitative outcomes, pain and quality of life in a randomised cross-over pilot trial of overnight supplementary oxygen and auto-adjusting continuous positive airways pressure (POMS2a): study protocol for a randomised controlled trial
BACKGROUND: Sickle cell anaemia (SCA) is an inherited disorder of haemoglobin. Patients experience long-term health care problems, affecting quality of life (QOL) including frequent acute pain, which is difficult to document in trials except as hospital admissions. Pilot data suggests that overnight respiratory support, either supplementary oxygen or auto-adjusting continuous positive airways pressure (APAP), is safe and may have clinical benefit. This pilot trial aims to determine which intervention is more acceptable to participants and whether there are other advantages of one over the other, e.g. in respiratory function or haematological parameters, before conducting the Phase 2 trial of overnight respiratory support funded by the National Institutes of Health Research.

METHODS/DESIGN: This is a pilot cross-over interventional trial with the order of interventions decided by simple randomization. Ten adults (age over 18 years) and 10 children (aged between 8 and 18 years) with homozygous sickle cell disease (haemoglobin SS, HbSS), recruited regardless of symptoms of sleep-disordered breathing, will undergo overnight pulse oximetry and will have two interventions, overnight oxygen and APAP, for a week each in randomised order with a washout week between interventions. Participants will complete online diaries via an iPad throughout the 29 days of the study and will complete QOL questionnaires and have measurement of haematology, biochemistry, spirometry and lung volumes (adults only) at 3 time points, at baseline and after each intervention, as well as in-depth semi-structured qualitative interviews after each intervention, carried out by an experienced psychologist. Both qualitative and statistical methods will be used to analyze the data. The primary outcome is qualitative data looking at participant experience from the transcribed interviews after each intervention. The participant's view on feasibility, acceptability and preference will specifically be explored. The QOL, laboratory and lung function data will be compared with baseline for each arm.

DISCUSSION: Patient and public involvement is an integral part of this trial and the key outcome is the qualitative result, which is dependent on obtaining good quality data to advise on participant feasibility, acceptability and preference. This is being addressed by using a standard interview. The development of a pain endpoint is another important outcome and collecting daily measurements is likely to be challenging. Research results will be used to inform design of the Phase 2 trial.

TRIAL REGISTRATION: ISRCTN46078697 18 July 2014.
sickle cell anaemia, inherited diseases, harmoglobin, qualitative method, statistical method, randomised controlled trial
1745-6215
1-11
Howard, Jo
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Inusa, Baba
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Liossi, Christina
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Jacob, Eufemia
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Murphy, Patrick B.
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Hart, Nicholas
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Gavlak, Johanna
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Sahota, Sati
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Chorozoglou, Maria
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Nwosu, Carol
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Gwam, Maureen
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Gupta, Atul
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Rees, David C.
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Thein, Swee Lay
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Reading, Isabel C.
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Kirkham, Fenella J.
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Cheng, Man Yeung Edith
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Howard, Jo
6c7512d5-7a3d-4a5c-b874-19c83eab11d2
Inusa, Baba
830cfc6c-2482-4986-b2d3-9edd5cf2d9f5
Liossi, Christina
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Jacob, Eufemia
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Murphy, Patrick B.
6ec55e2b-bf7c-47fc-9530-06be8252c1ba
Hart, Nicholas
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Gavlak, Johanna
15bfdfeb-374b-4108-86c8-ec0a1dade98e
Sahota, Sati
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Chorozoglou, Maria
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Nwosu, Carol
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Gwam, Maureen
07be3563-c091-41d5-ab22-9776aee02996
Gupta, Atul
ef653ca8-50c8-4aab-a094-68e8c9879242
Rees, David C.
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Thein, Swee Lay
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Reading, Isabel C.
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Kirkham, Fenella J.
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Cheng, Man Yeung Edith
527314dd-0f74-4d4e-85d6-56e5d3c04584

Howard, Jo, Inusa, Baba, Liossi, Christina, Jacob, Eufemia, Murphy, Patrick B., Hart, Nicholas, Gavlak, Johanna, Sahota, Sati, Chorozoglou, Maria, Nwosu, Carol, Gwam, Maureen, Gupta, Atul, Rees, David C., Thein, Swee Lay, Reading, Isabel C., Kirkham, Fenella J. and Cheng, Man Yeung Edith (2015) Prevention of morbidity in sickle cell disease-qualitative outcomes, pain and quality of life in a randomised cross-over pilot trial of overnight supplementary oxygen and auto-adjusting continuous positive airways pressure (POMS2a): study protocol for a randomised controlled trial. Trials, 16 (376), 1-11. (doi:10.1186/s13063-015-0883-y). (PMID:26303626)

Record type: Article

Abstract

BACKGROUND: Sickle cell anaemia (SCA) is an inherited disorder of haemoglobin. Patients experience long-term health care problems, affecting quality of life (QOL) including frequent acute pain, which is difficult to document in trials except as hospital admissions. Pilot data suggests that overnight respiratory support, either supplementary oxygen or auto-adjusting continuous positive airways pressure (APAP), is safe and may have clinical benefit. This pilot trial aims to determine which intervention is more acceptable to participants and whether there are other advantages of one over the other, e.g. in respiratory function or haematological parameters, before conducting the Phase 2 trial of overnight respiratory support funded by the National Institutes of Health Research.

METHODS/DESIGN: This is a pilot cross-over interventional trial with the order of interventions decided by simple randomization. Ten adults (age over 18 years) and 10 children (aged between 8 and 18 years) with homozygous sickle cell disease (haemoglobin SS, HbSS), recruited regardless of symptoms of sleep-disordered breathing, will undergo overnight pulse oximetry and will have two interventions, overnight oxygen and APAP, for a week each in randomised order with a washout week between interventions. Participants will complete online diaries via an iPad throughout the 29 days of the study and will complete QOL questionnaires and have measurement of haematology, biochemistry, spirometry and lung volumes (adults only) at 3 time points, at baseline and after each intervention, as well as in-depth semi-structured qualitative interviews after each intervention, carried out by an experienced psychologist. Both qualitative and statistical methods will be used to analyze the data. The primary outcome is qualitative data looking at participant experience from the transcribed interviews after each intervention. The participant's view on feasibility, acceptability and preference will specifically be explored. The QOL, laboratory and lung function data will be compared with baseline for each arm.

DISCUSSION: Patient and public involvement is an integral part of this trial and the key outcome is the qualitative result, which is dependent on obtaining good quality data to advise on participant feasibility, acceptability and preference. This is being addressed by using a standard interview. The development of a pain endpoint is another important outcome and collecting daily measurements is likely to be challenging. Research results will be used to inform design of the Phase 2 trial.

TRIAL REGISTRATION: ISRCTN46078697 18 July 2014.

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Accepted/In Press date: 22 July 2015
Published date: 25 August 2015
Keywords: sickle cell anaemia, inherited diseases, harmoglobin, qualitative method, statistical method, randomised controlled trial
Organisations: Clinical & Experimental Sciences

Identifiers

Local EPrints ID: 390150
URI: http://eprints.soton.ac.uk/id/eprint/390150
ISSN: 1745-6215
PURE UUID: 5a12ba29-c1be-491c-975e-29f46f48a81c
ORCID for Christina Liossi: ORCID iD orcid.org/0000-0003-0627-6377
ORCID for Isabel C. Reading: ORCID iD orcid.org/0000-0002-1457-6532
ORCID for Fenella J. Kirkham: ORCID iD orcid.org/0000-0002-2443-7958

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Date deposited: 21 Mar 2016 13:36
Last modified: 15 Mar 2024 03:24

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Contributors

Author: Jo Howard
Author: Baba Inusa
Author: Eufemia Jacob
Author: Patrick B. Murphy
Author: Nicholas Hart
Author: Johanna Gavlak
Author: Sati Sahota
Author: Maria Chorozoglou
Author: Carol Nwosu
Author: Maureen Gwam
Author: Atul Gupta
Author: David C. Rees
Author: Swee Lay Thein
Author: Isabel C. Reading ORCID iD
Author: Man Yeung Edith Cheng

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