ISSN 2312-5705
Volume 1 Issue 2, December 2014
MJHS
MALTA JOURNAL OF HEALTH SCIENCES
FACULTY OF HEALTH SCIENCES
www.um.edu.mt/healthsciences/mjhs
Malta Journal of Health Sciences - Journal of the Faculty of Health Sciences
https://www.um.edu.mt/healthsciences/mjhs/
mjhs@um.edu.mt
Editorial Board
Chairperson
Professor Angela Xuereb
Dean, Faculty of Health Sciences,
University of Malta,
MSD 2080, Malta.
angela.a.xuereb@um.edu.mt
Editor-in-Chief
Dr Daniela Gatt
Department of Communication Therapy,
University of Malta,
MSD 2080, Malta.
mjhs@um.edu.mt
Associate Editors
Dr Stephen Lungaro-Mifsud
Department of Physiotherapy
stephen.lungaro-mifsud@um.edu.mt
Dr Josianne Scerri
Department of Nursing
josianne.scerri@um.edu.mt
Dr Victoria Sultana
Department of Nursing
victoria.sultana@um.edu.mt
Dr Vasilis Valdramidis
Department of Food Studies & Env. Health
vasilis.valdramidis@um.edu.mt
Dr Francis Zarb
Department of Radiography
francis.zarb@um.edu.mt
Advisors
Dr Rita Borg Xuereb, Department of Midwifery
Dr Sandra Buttigieg, Department of Health Services Management
Dr Cynthia Formosa, Department of Podiatry
Prof. Carmel J. Caruana, Department of Medical Physics
Mr René Mifsud, Department of Occupational Therapy
Copy Editors
Web Administrators
Dr Stephen Lungaro-Mifsud
Dr Vasilis Valdramidis
Dr Francis Zarb
Ms Juanita Briffa
juanita.briffa@um.edu.mt
Mr Nicolai Schembri
nicolai.schembri@um.edu.mt
Aim and scope
The Malta Journal of Health Sciences is a peer-reviewed, open
access publication that promotes the sharing and exchange of
knowledge in Health Sciences. It provides a platform for novice
and established researchers to share their findings, insights and
views within an inter-professional context. The Journal originates
within the Faculty of Health Sciences, University of Malta.
The Malta Journal of Health Sciences disseminates research on
a broad range of allied health disciplines. It publishes original
research papers, review articles, short communications, commentaries, letters to the editor and book reviews. The readership of
the journal consists of academics, practitioners and trainee health
professionals across the disciplines of Applied Biomedical Science,
Audiology, Communication Therapy, Community Nursing, Environmental Health, Food Science, Health Services Management,
Medical Physics, Mental Health Nursing, Midwifery, Nursing, Occupational Therapy, Physiotherapy, Podiatry and Radiography.
Submitted manuscripts undergo independent blind peer review, typically by two reviewers with relevant expertise. All
manuscripts are reviewed as rapidly as possible and an editorial
decision is generally reached within approximately two months
of submission. Authors of manuscripts that require revisions
will have two weeks to submit their revised manuscripts. No
manuscript that has already been published or is under consideration for publication elsewhere will be considered.
Types of manuscripts
Review articles will present a substantial survey with an
adequate historical perspective of the literature in an area of
Health Sciences. The manuscript should not exceed the limit of
5,000 words.
Research papers should be manuscripts comprising complete
reports of original, scientifically sound research. They must
contribute new knowledge, be prepared for a wide readership and
should not exceed 4,000 words.
Short communications should be brief reports of scientifically
sound research, but of limited scope e.g. limited amount of data,
that contribute new knowledge. They should not exceed 2,000
words.
Commentaries should be focused and opinionated articles on
any subject within the scope of the Journal. These articles
should be related to a contemporary issue, such as recent research
findings. They should not exceed 2,000 words.
Letters to the editor should provide further debate on a topic
addressed by a paper published in the Journal. Authors of the
manuscript will be sent an edited copy of the letter and they will
have the right to reply. They should not exceed 1,000 words.
Book reviews should be timely and objective. They should
discuss and assess the main ideas and major objectives of
the book and how effectively these are accomplished. Finally,
constructive comments about the strengths and weaknesses of the
book should be presented. They should not exceed 1,000 words.
The manuscript lengths cited above do not include the Abstract
and References.
Instructions for authors
Prior to manuscript submission, it should be ensured that the
following guidelines are adhered to. Manuscripts not conforming
to these guidelines may be returned.
Submission of Manuscripts
Manuscripts are to be emailed to the Editor-in-Chief on
mjhs@um.edu.mt
General guidelines
All papers must be written in correct and consistent English
(UK). The manuscript, including references, tables and figure
legends, should be typed in double line spacing, with margins at
least 25 mm on each side. Text should be standard 10 or 12 pt.
Author names and/or affiliations should not appear anywhere
in the manuscript pages or images in order to ensure blind peer
review. Continuous line numbers should be added to the whole
document (in Microsoft Word: View menu → Print Layout →
Layout → Text Layout → Line Numbers).
Title page: The full title page should also be submitted as a
separate document and should include the following:
Title of paper Abbreviations should not be used in the title.
Submission category This should be specified as full research
paper, book review etc.
Authors Full names of all authors are to be provided, followed
by qualifications, affiliations and addresses. The corresponding
author should be indicated and full details, including email and
mailing addresses and telephone numbers, should be provided.
Running Head A running headline of not more than 40
characters should be included at the top of the page.
Blind title page: A blind title page giving only the title without
author details should also be submitted.
Abstract: The abstract should be an accurate summary of the
paper presented in one paragraph (not exceeding 300 words).
The abstract should:
(i) indicate the specific purpose of the article;
(ii) describe the methodology used to achieve the purpose;
(iii) present the findings including key statistics (this section
may be omitted for articles that are not data-based);
(iv) present the conclusions based on the data provided;
(v) highlight the novelty of the work.
The abstract should not include references.
Keywords: Immediately after the abstract, a list of up to 6 key
words is to be included. Selected words and phrases should relate
directly to the content of your work.
N.B. The title, abstract and keywords should be chosen carefully
as they determine the ease with which readers access the article
through online search engines.
Main text: The article text should consist of the sections
outlined below, in the same order. Results or Findings and
Discussion could also be combined. Repetitions between sections
should be avoided. Abbreviations and acronyms may be used
where appropriate but must always be defined at their first
mention.
Introduction Briefly describe the purpose of your work, explain
its importance and provide a relevant theoretical background.
Methods Describe the research design, the materials and/or
participants and the methods used for data collection. Sufficient
detail should be provided to allow reproduction of the study
design.
Results or Findings Present results in a clear and logical
sequence. If tables are used, do not duplicate the data in the text
but describe trends and main points.
Discussion This should provide an interpretation of the results
without repeating them.
Conclusions The main conclusions of the paper are to be
presented in this section.
Acknowledgements (if relevant) Collate acknowledgements
in a separate section at the end of the article before the references.
N.B. Please refer to the section on Ethical Guidelines for more
information.
References Authors are responsible for the accuracy of references. All references must appear both in the text and the
reference list. References should follow the Harvard referencing
system. In the text, the information required is:
the surname of the authors;
the year of publication;
the page number (direct quotes only).
Commas are used to separate information within parentheses
e.g. (author’s name, year of publication). Full stops are always
used after the abbreviation p. for page number e.g. p.34 (or pp.
for multiple pages). If the citation is at the end of the sentence, it
is considered to be part of the sentence, so the full stop is placed
after the closing parenthesis.
Examples
Book (1 author)
Neville, C. (2010) The Complete Guide to Referencing and
Avoiding Plagiarism. 2nd Ed. Maidenhead: Open University
Press.
In text:
Neville (2010) argues that...
Book (2 to 3 authors)
Bradbury, I., Boyle, J. & Morse, A. (2002) Scientific Principles
for Physical Geographers. Harlow: Prentice Hall.
In text:
Bradbury, Boyle and Morse (2002)...
Book (4 or more authors)
Reece, J.B., Urry, L.A., Cain, N.L., Wasserman, S.A., Minorsky,
P.V. & Jackson, R.B. (2012) Campbell Biology. 9th Ed. London:
Benjamin Cummings.
In text:
(Reece et al., 2012)....
Journal article (printed)
Trefts, K. & Blacksee, S. (2000) Did you hear the one about
Boolean Operators?
Incorporating comedy into the library
induction. Reference Services Review, 28(4), pp.369-378.
In text:
Trefts and Blacksee (2000) argue that....
Journal article (electronic/online)
Wilson, J. (1995) Enter the Cyberpunk librarian: future directions in cyberspace. Library Review. [Online] Emerald Database
44(8), pp.63-72. Available from: http://www.emeraldinsight.com.
[Accessed: 30th January 2012].
In text
Wilson (1995) argues that.....
Website
BBC News.(2008)Factory gloom worst since 1980. [Online] Available
from:http://news.bbc.co.uk/1/hi/business/7681569.stm.
[Accessed: 19th June 2012].
References should be arranged first alphabetically and then
further sorted chronologically if necessary.
More than one
reference from the same author(s) in the same year must be
identified by the letters “a”, “b”, “c” etc., placed after the year of
publication.
Tables should be referred to specifically in the text of the article
and provided in the same document. They should be in editable
MS Word format.
Number tables consecutively with Arabic numerals (1, 2, 3
etc.), in the order in which they appear in the text. Each table
should have a short descriptive title, be self-explanatory and
not duplicate data given in the text or figures. Ensure that all
columns and rows are properly aligned. Include horizontal rules
at the top and bottom of a table and one below the column headings. If a column heading encompasses two or more subheadings,
then the main headings and subheadings should be separated by
a single short rule. No other rules should be included, neither
horizontal nor vertical. Appropriate space should be used to
separate columns. Rows should be double-spaced.
A table may have footnotes if necessary. Each footnote should
begin on a new line. Abbreviations in tables should be defined in
footnotes even if defined in the text or in a previous table.
Figures should be referred to specifically in the text of the
paper. Number figures consecutively using Arabic numerals and
avoid repeating material in the text. Abbreviations used in
figures should be defined in the caption.
Image files should be supplied in EPS, TIFF, PDF, PPT
or JPEG format, with each figure uploaded separately and
numbered. Unless essential to the content of the article, all
illustrations should be supplied in black and white with no colour
contained within them. Files should be saved at the appropriate
dpi (dots per inch) for the type of graphic - definitely not less
than the typical screen value of 72 dpi for both the vertical and
horizontal directions. Lower resolutions will not be usable.
Save line drawings at 800 dpi or 1200 dpi for fine line work
and 300 dpi for halftone and colour work. The image should
be cropped to show just the relevant area, while the amount of
white space around the illustration should be kept to a minimum.
Supply illustrations at the size they are to be printed, usually 76
mm wide (single column of text) or, for especially large figures,
161 mm wide (two columns of text). All annotations should be
included within the images supplied.
Participant identity must be obscured.
Additional points to note:
(i) do not put a box around graphs, diagrams or other artwork;
(ii) avoid background gridlines unless these are essential (e.g.
confidence limits);
(iii) ensure that lettering is appropriately sized and corresponds
to 8 or 9 pt when printed;
(iv) include all units of measurement on axes;
(v) all lines (e.g. graph axes) should have a minimum width
of 1/4 pt (0.1 mm), otherwise they will not print; 1 pt weight is
preferable;
(vi) avoid using tints (solid black and white or variations of
crosshatching are preferred). Any tints that are used must be at
a minimum 5% level to print (but do not use too high a tint as it
may print too dark);
(vii) do not use three-dimensional histograms when the
addition of a third dimension gives no further information.
Appendices are discouraged if the material can be included in
the main text. If an appendix is necessary e.g. mathematical
calculations that would disrupt the text, it should be supplied
as a separate file. If more than one appendix are included, these
should be identified using different letters. An appendix may
contain references. Appendices must be referred to in the main
text.
Footnotes should be used sparingly. If used, they should be
numbered consecutively throughout the paper using superscript
Arabic numbers. Do not include footnotes in the reference list.
Peer review process
All manuscripts are pre-screened by the Editor-in-Chief (or
Editorial Board member) upon submission. Priority is given to
articles that will help allied health professionals to make better
decisions, increase pertinent knowledge base and support high
quality research. Immediate manuscript rejection by the Malta
Journal of Health Sciences is expected in the following instances:
(i) study does not have a theoretical or evidence-based
argument for the relevance of the work to allied health practice;
(ii) manuscript does not contain sufficiently new or important
information and therefore does not offer a significant contribution
to the literature;
(iii) reliability study does not include a discussion of the
influence that the findings will have in the field;
(iv) study evaluates a new test without a sound comparison to
current tests;
(v) study has serious flaws in the Methods section;
(vi) authors did not obtain ethics approval from a properly
constituted ethics committee, where appropriate.
Manuscripts that qualify for review are evaluated by at least two
experts as appropriate. A double blind review process is adopted.
After the reviewers receive a paper from the editor, they read it
closely and provide individual critiques in which they:
(i) comment on the validity of the science, identifying scientific
errors and evaluating the design and methodology used;
(ii) judge its significance by evaluating the importance of the
findings;
(iii) determine the originality of the work based on how much
it advances the field;
(iv) recommend that the manuscript is rejected or accepted
with minor revision; major revision will entail resubmission and
further review.
The Editor-in-Chief/Editorial Board member reads the
manuscript and the reviewers’ comments, makes his/her own
comments, and provides guidance to the Editorial Board on the
suitability of the manuscript for publication. The Editor-in-Chief
does not need to heed this recommendation. The Editorial Board
will then decide on the content of the feedback letter to the
author/s.
Ethical guidelines
The Malta Journal of Health Sciences adheres to the following
ethical guidelines for research and publication.
Authorship
The definition of authorship is based on the following criteria:
(i) significant contribution of authors to the conception and
design of the research, data collection or analysis and interpretation of data;
(ii) drafting and/or critically reviewing the manuscript for its
academic content;
(iii) providing final approval of the version to be submitted (or
resubmitted) for publishing.
Authors are expected to meet all three criteria. Contributors who
do not qualify under the authorship criteria should be included
in the Acknowledgments section.
Acknowledgments
Any acknowledgements should be included at the end of the
article, prior to the Declaration of Conflicting Interests (if applicable) and your References. Any sources of funding must also be
acknowledged under a separate heading entitled Funding, directly
after the Acknowledgments section. Funding acknowledgments
should be in the form of a sentence as shown below, with the
funding agency written in full, followed by the grant number in
square brackets:
This work was supported by XXX [grant number xxx].
Declaration of conflicting interests
Authors are expected to disclose any associations that might
pose a conflict of interest in connection with the submitted
manuscript. All sources of funding or products must be included
in the Acknowledgements section of the manuscript.
Originality of work
Submission of a manuscript implies that the work described:
(i) has not been published before (except in the form of an
abstract or academic thesis), and
(ii) is not under consideration for publication elsewhere. If the
manuscript is accepted for publication, it cannot be published
in the same form elsewhere, in print or electronically, in English
or in any other language, without the written consent of the
copyright-holder.
Permissions
When previously published material (e.g. tables, photographs
or illustrations) are included in submitted manuscripts, the
authors are required to obtain a written statement from the
copyright holder giving permission for reproduction. A copy of
the permission obtained must be submitted to the Editor-in-Chief
of this journal.
Author misconduct and article withdrawal
As a policy, submitted articles are checked for plagiarism using
duplication detecting software. The Editor-in-Chief reserves the
right to reject any manuscript where it is suspected there is
unethical conduct during a research project, or in the manuscript
authoring or content.
Studies involving human and animal subjects (details)
Research undertaken with human and animal subjects
must have been carried out in accordance with relevant
internationally accepted guidelines such as the Declaration of Helsinki for clinical research involving humans
(http://www.wma.net/en/30publications/10policies/b3/
index.html), recommendations by the International committee of Medical Journal Editors (http://www.icmje.org), the
British Educational Research Association ethical guidelines
(http://www.bera.ac.uk/publications/Ethical%20Guidelines)
and the EU Directive 2010/63/EU for research on animals
(http://ec.europa.eu/environment/chemicals/lab_animals/
legislation_en.htm).
Ethical approval
Ethical approval from a research ethics committee and/or institutional review board is usually required and details regarding
the board providing approval should be listed in the Methods
section of the manuscript.
Publishing policy
Copyright on the published article will be retained by the
author/s. Authors grant the Malta Journal of Health Scienceslicense to publish the article and identify itself as the
original publisher. In addition, authors may allow any third party
the right to use the article as long as its original authors, citation
details and publisher are identified, save for commercial purposes.
Individual users may access, download, copy, display and
redistribute to colleagues open access Malta Journal of Health
Sciences articles solely for non-commercial and non-promotional
purposes.
Table of Contents
Malta Journal of Health Sciences Volume 1 - Issue 2 (December 2014)
Editorial
28
Editorial
Daniela Gatt
Regular Articles
29
Key environmental stress biomarker candidates for the optimisation of chemotherapy treatment of leukaemia
Review Article
Eirini G. Velliou, Susana Brito Dos Santos, Maria Fuentes-Garí, Ruth Misener, Eleni Pefani, Nicki Panoskaltsis,
Athanasios Mantalaris and Efstratios N. Pistikopoulos
35
Degree of burnout among Maltese podiatrists working in two different settings Research Article
Trista Agius and Cynthia Formosa
40
The effectiveness of social communication groups in mainstream schools Research Article
Tiziana Baldacchino and Kristina Agius
54
The lived experience of persons with an amputation Research Article
Chanelle Grech and Roberta Farrugia Debono
60
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies Research Article
Martina Galdes and Daniela Gatt
Malta Journal of Health Sciences
http://www.um.edu.mt/healthsciences/mjhs
DOI: http://dx.medra.org/10.14614/EDITLANG.2.28
Editorial
Daniela Gatt
Editor-in-Chief, Malta Journal of Health Sciences
As we launch Issue 2 of the Malta Journal of Health Sciences
(MJHS), I would like to share with you some thoughts on one particular topic which I hold very much at heart, namely children’s
language development. In particular, I would like to highlight the
importance of social interaction in helping children to develop into
effective communicators.
The complex process of language learning is grounded in the
child’s social experiences. In the months preceding the emergence
of language, babies thrive on the attention, smiles, language input and physical contact of their caregivers. Early communicative
exchanges drive them to channel their reflexive actions and vocalisations into purposeful ones. In early language learning, the child
depends on social interactions with significant others to be able to
forge links between the speech sounds and meanings they receive
in their input. Language needs to be experienced naturally, as it
is used to identify and describe entities, persons, objects and experiences in daily settings. Interactive experiences help the child
to crack the language code and realise the value of language in
enabling a wide range of communicative functions. As children’s
language capacity expands, communicative exchanges allow them
to practise available language skills and develop new ones.
Experiencing the communicative potential of language is priceless for language-learning children. It is not merely a question
of exposing the child to the ambient language. While technology
may assist the child’s language learning, it is no substitute for oneto-one interactions in encouraging the uptake of language. Strikingly, children are extremely adept at using technological devices
and applications, to the extent that they often become engrossed
in them and ignore other forms of input. Although such devices
can be useful and educational in their own right, they surely cannot make up for the rich language input that may be derived from
meaningful one-to-one or group interactions. These not only support language development but also assist the learning of social
rules for communication. The sheer beauty and effectiveness of
simple, face-to-face exchanges tends to be under-estimated. Communicative exchanges in everyday contexts provide children with
the necessary tools for learning language and developing social
skills. Through social interactions, infants and toddlers learn the
basic components of language and test their newly-acquired skills
in real-life settings. Involvement in social exchanges enables older
children to learn the rules for social language use and to practise
organising multiple thoughts in a coherent sequence prior to verbalising them. Participation in conversations equips them with
skills for understanding and using shades of humour and sarcasm,
among others. Importantly, they also develop an appreciation
of others’ perspectives and emotions, while developing their own.
These are but a few of the advantages that may be derived from
encouraging children’s involvement in social exchanges.
In today’s day and age, isolation from face-to-face communication has become acceptable. However, we cannot afford to have
our children detach themselves from social exchanges. To reap the
full benefits of the technological facilities available today, they can
be focused on and explored in interactional exchanges. Children
can be encouraged to share, describe and discuss their favourite
gadgets, programmes and applications. This should go a long
way in encouraging them to appreciate the social dimension of
language and become truly effective communicators.
Having aired my views on the importance of social engagement
for children’s language and communication development, I now invite you to explore the articles in this issue. The diversity of topics
should make for interesting and intriguing reading. I trust that
these papers will spur us on to engage in further health science
research.
Malta Journal of Health Sciences Online
http://www.um.edu.mt/healthsciences/mjhs
DOI: http://dx.medra.org/10.14614/CHEMOLEUK.2.29
Review paper
KEY ENVIRONMENTAL STRESS BIOMARKER CANDIDATES
FOR THE OPTIMISATION OF CHEMOTHERAPY TREATMENT
OF LEUKAEMIA
Eirini G. Velliou1 , Susana Brito Dos Santos2 , Maria Fuentes-Garí2,3 , Ruth Misener4 , Eleni Pefani3 ,
Nicki Panoskaltsis5 , Athanasios Mantalaris2 , Efstratios N. Pistikopoulos3
1
Department of Chemical and Process Engineering, Faculty of Engineering and Physical Sciences, University of Surrey, Guildford, Surrey, GU2 7XH, U.K.
2
Biological Systems Engineering Laboratory (BSEL), Department of Chemical Engineering, Imperial College
London, South Kensington Campus, London, U.K.
3
Centre for Process Systems Engineering (CPSE), Department of Chemical Engineering, Imperial College London,
South Kensington Campus, London, U.K.
4
Department of Computing, Imperial College London, South Kensington Campus, London, U.K.
5
Department of Haematology, Imperial College London, Northwick Park and St. Mark’s Campus, London, U.K.
Abstract. The impact of fluctuations of environmental parameters such as oxygen and starvation on the evolution of leukaemia
is analysed in the current review. These fluctuations may occur
within a specific patient (in different organs) or across patients
(individual cases of hypoglycaemia and hyperglycaemia). They
can be experienced as stress stimuli by the cancerous population,
leading to an alteration of cellular growth kinetics, metabolism
and further resistance to chemotherapy. Therefore, it is of high
importance to elucidate key mechanisms that affect the evolution
of leukaemia under stress. Potential stress response mechanisms
are discussed in this review. Moreover, appropriate cell biomarker
candidates related to the environmental stress response and/or
further resistance to chemotherapy are proposed. Quantification
of these biomarkers can enable the combination of macroscopic kinetics with microscopic information, which is specific to individual
patients and leads to the construction of detailed mathematical
models for the optimisation of chemotherapy. Due to their nature,
these models will be more accurate and precise (in comparison
to available macroscopic/black box models) in the prediction of
responses of individual patients to treatment, as they will incorporate microscopic genetic and/or metabolic information which is
patient-specific.
Keywords: leukaemia, environmental stress, hypoxia, autophagy, chemotherapy, cellular biomarkers
Correspondence to: E. G. Velliou
(e.velliou@surrey.ac.uk)
Received: 24.07.2014 - Revised: 25.09.2014 - Accepted: 14.10.2014
- Published: 17.12.2014
c 2014 The authors
1
Introduction
Leukaemia is a severe cancer of the haematopoietic system characterised by the incapability of blood progenitors to mature normally, leading to the accumulation of immature white blood cells,
the so-called blasts, in the bone marrow (BM) (Beutler et al.,
2001). Alternatively, this disease can be viewed as the formation
of an abnormal haematopoietic tissue, the initiation of which is the
result of the function of a small amount of Leukaemic Stem Cells
(LSCs) (Passegué et al., 2003). Depending on the decrease of the
normal blood cell populations, the disease symptoms can consist
of fatigue, haemorrhage, infections and fever. The health condition of the patient depends on the amount of normal blood cells
compared to the amount of those that are leukaemic. According to
Cancer Research U.K. (2014), 8,616 people in the U.K. were diagnosed with leukaemia in 2011, with 4,503 deaths from this disease
occurring in 2010. Moreover, 82,300 new cases of leukaemia were
diagnosed in 2012 within the European Union. Leukaemia can
be divided into different types, depending on the haematopoietic
lineage in which the proliferation disorder occurs. More specifically, myeloid leukaemia occurs in the myeloid lineage and lymphocytic leukaemia occurs in the lymphoid lineage. Depending
on the speed of evolution of the disease, leukaemia can be divided
into acute, whereby the number of blasts increases rapidly, leading
to a faster disease evolution, and chronic i.e. the progress of the
disease is slower as there is production of partly mature but not
fully functional white blood cells. Based on this categorisation,
the following four general types of leukaemia can be found: Acute
Myeloid Leukaemia (AML), Chronic Myeloid Leukaemia (CML),
Acute Lymphocytic Leukaemia (ALL) and Chronic Lymphocytic
Leukaemia (CLL).
Acute Myeloid Leukaemia (AML) is one of the most aggressive
types of leukaemia. According to Cancer Research U.K. (2014),
approximately 2,921 cases of AML occurred in 2011 in the U.K.
Key environmental stress biomarker candidates for the optimisation of chemotherapy treatment of
leukaemia
AML is a type of leukaemia which is characterised by an accumulation of immature blasts in the myeloid lineage and, as a consequence, insufficient blood cell production. These blasts have low
proliferation capability. However, a small population of cells with
higher proliferation capacity and self-renewal potential, the LSCs,
are the key component for maintenance of the disease (Bonnet
& Dick, 1997). AML is usually the result of somatic mutations
in either a pluripotent haematopoietic stem cell or in a slightly
differentiated progenitor cell, leading to a deregulation and/or inhibition of normal haematopoiesis due to space restrictions and
inhibitory and clonal factors specific to the disease (Lowenberg,
Downing & Burnett, 1999; Lichtman, 2001; Panoskaltsis, Reid &
Knight, 2003).
Chronic Myeloid Leukaemia (CML), alternatively known as
Chronic Granulocytic Leukaemia (CGL), is a stem cell disease of the myeloid lineage - characterised by an exaggerated granulocytosis, anaemia, granulocytic immaturity, basophilia and
splenomegaly. There is an extreme cellular accumulation in the
BM. From a genetic point of view, 90% of cases show a reciprocal translocation between chromosomes 9 and 22, the so-called
Philadelphia (Ph) chromosome translocation. In many cases, the
disease can progress into a very high speed phase, resembling AML
(Lichtman, 2001). CML is a rather rare cancer of the blood. According to Cancer Research U.K. (2014), 675 patients were diagnosed with CML in 2011 in the U.K.
Acute Lymphocytic Leukaemia (ALL) (alternatively known as
Acute Lymphoblastic Leukaemia) is a type of leukaemia which
mostly affects children. Approximately 4,000 cases of ALL are
reported on an annual basis in the U.S.A., of which two thirds
are children (Pui & Evans, 2006). According to Cancer Research
U.K. (2014), 654 cases of ALL were diagnosed in 2011 in the U.K.
ALL is a leukaemia in which the cellular deregulation occurs in
the lymphoid lineage of the haematopoietic system. More specifically, an abnormality in the lymphocytes takes place, leading to
an accumulation of non-functional white blood cells (blasts) in
the BM as a result of abnormal cellular proliferation, blocking of
cellular differentiation and increased resistance to apoptosis i.e.
cell death (Pui, 2009).
Chronic Lymphocytic Leukaemia (CLL) is a type of leukaemia
which is characterised by an accumulation of immature lymphocytes (of the B-cell lineage) in the human BM, peripheral blood
and the lymphoid tissues (Kipps, 2001). The progress of CLL is
much slower than that of ALL and it affects older adults i.e. people over 51 years of age. According to the National Health Service
(NHS) U.K., about 2,400 people in the U.K. are diagnosed with
CLL on an annual basis and the disease affects 2.7 persons per
100,000 in the U.S.A. (Kipps et al., 2001). According to Cancer
Research U.K. (2014), 3,233 people were diagnosed with CLL in
the U.K. in 2011.
The most common treatment for all types of leukaemia is
chemotherapy (Cancer Research U.K., 2014).
A variety of
chemotherapy drugs are generally used, depending on the type
of leukaemia. The most commonly used drugs for treatment of
leukaemia are cytarabine (cytosine arabinose or ara-C), which is
an antimetabolite targeting to block the DNA/RNA replication by
attacking the cells that are in the S-phase of the cell cycle, and the
anthracycline drugs (such as fludarabine) which attack cells that
are in the G1-phase of the cell cycle (American Cancer Society,
2013). Current chemotherapy treatment protocols are designed
on the basis of pre-clinical animal experiments and empirical clinical trials, as well as the acquired experience of specialist physicians. The design parameters for these protocols consist of the
patient BM aspirate examination (blasts percentage, immunophenotype, cytogenetic and molecular analysis) and physiological patient characteristics (height, weight) for the normalisation of the
dose applied on the body surface area (BSA).
http://dx.medra.org/10.14614/CHEMOLEUK.2.29
2
30
Towards
a
Personalised
Chemotherapy Treatment: integrating in vivo, in vitro and
in silico knowledge
As mentioned in the previous section, traditional clinical diagnosis and further treatment of leukaemia focuses on each patient’s
clinical symptoms and signs, as well as characteristics (such as sex
and family history) and laboratory imaging evaluation. This process is a reactive approach to the disease, initiating after the disease symptoms appear. Moreover, in the past, drug development
by pharmaceutical industries was based on empirical observations.
However, nowadays, with significant progress constantly taking
place in the areas of genomics, proteomics and metabolomics, it
is believed that specific information related to the genetic characteristics and proteomic and metabolomic profile of an individual
patient could be used for tailored medical care, or personalised
medicine. Some of the challenges in the delivery of personalised
medicine lie in (a) the fidelity and validity of current experimental, i.e. in vitro, systems used to investigate human disease, (b)
the integration of patient-specific and disease-specific datasets i.e.
in silico and (c) the application of these models in clinical practice
to identify simple targets and more efficient, yet less toxic, therapies for a specific condition, i.e. in vivo. Therefore, ’closing
the loop’ from in vivo to in vitro and in silico is a first
step towards optimisation and consequently, personalisation of chemotherapy treatment . Moving in that direction,
an appropriate platform has been developed at the Centre for Process Systems Engineering at Imperial College London, consisting
of three main blocks i.e. in vivo-in vitro-in silico which, upon
completion, will allow the prediction of optimal drug dosages for
each individual, based on specific (personalised) characteristics
(Velliou et al., 2014a). The main advantage of this platform is
the dynamic interaction of its three robust building blocks. As
opposed to existing computational or experimental studies, our
platform consists of the interaction of both: data from our in
vitro platform are used as an input for the development of mathematical tools. These mathematical tools are further validated
with patient data. Figure 1 describes the different parts of the
integrated platform that is currently being designed at the Centre for Process Systems Engineering at Imperial College London
(Velliou et al., 2014a). Development and optimisation of the in
vitro and in silico blocks will eventually bridge the gap between
laboratory experimentation, mathematical modelling and in vivo
optimal chemotherapy treatment for a specific individual.
A very innovative in vitro tool has been developed and is being used for the ex vivo long-term patient sample cultivation
at the Centre for Process Systems Engineering (Mortera-Blanco
et al., 2010). This tool is a three-dimensional highly porous
polyurethane-based matrix coated with collagen type I, therefore
mimicking the porosity as well as the extracellular matrix present
in the human BM. This three-dimensional system allows the longterm (up to 6 weeks) cultivation of patient cells in an environment
that mimics the in vivo one.
An in silico tool, or mathematical model, which enables the
estimation/design of chemotherapy protocols for a specific individual based on the personalisation of the drug schedule has been
developed by Pefani et al. (2013). This model consists of a Pharmacokinetic (PK) and a Pharmacodynamic (PD) part. The PK
part consists of mass balances of the drug distribution in different
body organs such as the heart, the liver and the BM. The PD
part calculates the effect of the drug on both normal and abnormal cells in the BM, which is the location of the tumour. The
model input is the treatment inflow, which is calculated based on
the drug administration route and the injection rate. The latter
is a function of body characteristics such as height and weight.
http://www.um.edu.mt/healthsciences/mjhs
Key environmental stress biomarker candidates for the optimisation of chemotherapy treatment of
leukaemia
Figure 1: Towards optimisation/personalisation of chemotherapy
for leukaemia treatment (adapted from Velliou et al., 2014a).
For cases in which the initial drug dose proposed clinically failed
to eliminate the cancerous population, an optimisation process
was followed in order to re-determine the optimal drug dose on
the basis of clinically relevant constraints. Remarkably, the model
output treatments suggest drug dosages similar to those used clinically but, for example, the scheduling is different. The optimised
treatment would have much better outcomes with respect to the
elimination of the tumour (Pefani et al., 2014).
Furthermore,
a more detailed cell cycle model based on the work of GarcíaMünzer et al. (2013, 2014) has been developed, consisting of a
multi-stage population balance model (MS-PBM) (Fuentes-Garí
et al., 2014). This model is distributed on cell cycle progressrelated events i.e. cyclin or DNA expression (different cyclins are
produced at different phases of the cell cycle and DNA is produced
at the S-phase of the cell cycle). Cell cycle kinetics are tracked
not only across different cell cycle phases but also within each
phase, thus allowing a more efficient monitoring of the evolution
of the leukaemic population throughout the cell cycle. The latter is of high importance for chemotherapy optimisation since, as
mentioned previously, most chemotherapeutic drugs are cell cyclespecific, targeting cells that are present in specific phases of the
cell cycle. This PBM has been successfully validated with experimental data (Fuentes-Garí, 2014).
Moving towards the delivery of optimal chemotherapy protocols for each individual, it is important to understand and further
incorporate in the mathematical models the impact of environmental stress. More specifically, the effect of alterations of environmental parameters (oxygen, temperature, nutrients) on the
evolution and further response of leukaemia to chemotherapy have
to be incorporated (Velliou et al., 2014a, b). Fluctuations of those
parameters can take place either across different patients or within
the same patient. For example, glucose levels in the blood can
differ across a diabetic, a hypoglycaemic and a normal patient.
During chemotherapy treatment, it is possible that a patient may
have fever; high body temperature can be experienced as a temperature shock by the cancerous population. Oxygen levels can
fluctuate in different body compartments e.g. the BM and peripheral blood. The impact of those environmental factors on the
leukaemic evolution is further discussed in the following section.
2.1
Environmental Stress and
Leukaemia
Fluctuations in the micro-environmental conditions of the BM
i.e. oxygen concentration, composition and concentration of nu-
http://dx.medra.org/10.14614/CHEMOLEUK.2.29
31
trients such as glucose, cytokines or other growth factors, may
be experienced as an environmental stress. As a consequence,
these fluctuations can highly affect the normal and abnormal
haematopoietic proliferation, metabolic activity as well as drug
resistance and further evolution. For example, several researchers
have shown that oxidative stress i.e. the increase in the concentration of Reactive Oxygen Species (ROS), leads to activation of
survival pathways and is a key factor that promotes progression of
cancerous stem cells as well as resistance to chemotherapy (see as
examples Adbal Dayem et al., 2010; Fruehauf & Mayskens, 2013;
Liu et al., 2009; Lyu et al., 2008).
Especially in the case of the abnormal haematopoietic situation
of leukaemia, alterations of the oxygen and glucose concentration
in the different body compartments e.g. in the BM and the peripheral blood or the liver, and, on the other hand, between patients,
i.e. individual cases of hypoglycaemia or hyperglycaemia, may
lead to a different stress adaptation of the leukaemic population.
The latter will most likely affect the cancer growth and inactivation kinetics, as well as the response to a chemotherapeutic drug
in vivo.
A variety of research studies have revealed the strong relation
between resistance i.e. longer survival and increased proliferation of haematopoietic and/or leukaemic cells and/or resistance
to chemotherapy, with (1) oxygen or (2) starvation stress in
vitro and/or in vivo. For example, Fecteau et al. (2013) observed
an increased in vitro survival of cells from BM aspirates of patients with CLL in 5% O2 compared to 20% O2 . This increased
survival under hypoxic conditions was a result of the Mesenchymal
Stromal Cells (MSCs’) increased proliferation and the production
of soluble pro-survival factors i.e. CXCL12. Interactions between
CLL and (increased) MSCs lead to enhanced CLL resistance.
Wilkinson, Tome and Briehl (2012) state that chronic oxidative stress may contribute to increased resistance of lymphoma
patients to chemotherapy. This is due to the fact that oxidative
stress leads to alteration of the mitochondria i.e. release of intermembrane proteins, which leads to an increased permeabilisation
of the outer mitochondrial membrane and further resistance to
apoptosis.
Zhou et al. (2010) pointed out the possible relation between
the relapse of AML and increased oxidative stress in vivo. Specifically, parameters related to oxidative stress e.g. activities of
adenosine deaminase and xanthine oxidase, antioxidant capacity
(T-AOC), levels of human thioredoxin (TRX) and indoleamine
2,3-dioxygenase, as well as expression of specific genes related to
oxidative stress, were monitored in patients with AML for a time
period between a primary and a relapsed status. Low T-AOC
and up-regulated TRX expression led to a relapse of the disease,
indicating a strong correlation between oxidative stress and AML
development and relapse.
Lodi et al. (2011) observed that hypoxia is a key factor that
affects metabolic activity i.e. adaptation of phospholytic and glycolytic metabolism, and evolution of KG1a and K562 leukaemic
cell lines. Mitochondrial respiration remained unaltered for both
cell lines, indicating the ability of these leukaemic cell lines to increase their resistance under oxidative stress.
Giuntoli et al. (2011) studied the effect of the level of glucose
on the growth and proliferation of K562 cell lines, U937 cell lines
or primary CML cells under hypoxic conditions i.e. 0.1% O2 , as
well as under normoxia i.e. 21% O2 . Although in general, slower
growth was observed for lower glucose concentrations in hypoxia
as well as in normoxia, glucose shortage in hypoxia led to increased size of the leukaemic population compared to the normal
haematopoietic one.
Herst et al. (2011) have pointed out a possible relation between
the level of glycolytic metabolism of AML blasts and resistance
to chemotherapy. Analysis of 26 BM aspirates showed that AML
cells with higher glucose consumption were more tolerant to in
vitro apoptosis caused by ATRA and/or ATO drug.
http://www.um.edu.mt/healthsciences/mjhs
Key environmental stress biomarker candidates for the optimisation of chemotherapy treatment of
leukaemia
All previously mentioned studies point out that there is a
very strong relation between oxidative or starvation stress and
the leukaemic evolution i.e. growth, kinetics or resistance to
chemotherapy. Therefore, the possible oxidative and starvation
cellular stress should be systematically followed and/or quantified experimentally (in vitro) and eventually further incorporated
in mathematical models (in silico). Efficient monitoring of the
level of oxidative and metabolic stress and further adaptation can
take place via the selection and further quantification of specific
biomarker(s) i.e. intracellular molecules, the expression and/or
concentration of which may alter depending on the fluctuation
of oxygen and glucose in the system. In order to select ‘stress’
biomarker(s), an in-depth investigation of possible mechanisms of
oxidative and starvation stress cellular responses is needed (Velliou et al., 2014a). Hereafter, autophagy, a crucial mechanism
which is activated in the absence of nutrients and low oxygen levels, is described and potential biomarker molecules related to the
cell stress response are summarised.
2.2
(Macro-)Autophagy: the cellular response to metabolic stress and hypoxia
Autophagy is a cellular mechanism which aims at the maintenance of homeostasis of a normal cell, via degradation of organelles
and cellular components by the lysosomes (Banerji & Gibson,
2012; Choi, Ryter & Levine, 2013; Kongara & Karantza, 2012,
Levine & Kroemer, 2008; Lozy & Karantza, 2012). Autophagy
is activated as a result of exposure to a stress factor and most
probably in the absence of nutrients i.e. glucose starvation, as
well under hypoxic conditions (Lum et al., 2005; Scherz-Shouval
et al., 2007). Degradation of damaged mitochondria as well as
aggregation of proteins and other cellular organelles via the autophagic mechanism protect the cells from apoptotic signalling
(Jin & White, 2007; Moore, Allen & Sommerfield, 2006). Autophagy may be important in the regulation of cancer development and in the determination of the response of cancer cells to
chemotherapy (Degenhardt et al., 2006; Hippest, O’Toole & Thorburn, 2006; Wilkinson, Tome & Briehl, 2012).
Several researchers have shown that autophagy plays a crucial role in maintenance of normal haematopoiesis and function of
Haematopoietic Stem Cells (HSCs) (Kundu et al., 2008; Warr et
al., 2013). Others have shown that autophagy may lead to an increased resistance to chemotherapy and survival of several cancers,
including haematological malignancies. For example, Mortensen,
Watchon & Simon (2011) observed that loss of autophagy resulted
in loss of normal function of murine HSCs, leading to the expansion of a progenitor cell population in the BM which has as a
consequence a severe myeloproliferation. This myeloproliferation
strongly resembled human AML, indicating a possible link between maintenance of autophagy and avoidance of malignancies
such as AML. Wallington-Beddoe et al. (2011) showed that activation of autophagy (induced by the FTY720 drug) leads to
increased survival of ALL cells.
2.3
2.3.1
Biomarker Candidates for the Prediction of the Disease Evolution under Stress
(Autophagic) Biomarker candidates
For ‘switching on’ and maintenance of the autophagic response,
a variety of genes are over-expressed and proteins, mainly
kinases, are activated and/or de-activated, depending on whether
they have a positive or negative regulatory role in autophagy.
Therefore, in order to monitor autophagy in an in vitro system,
many different biomarkers of genomic and/or protein level can
be considered (see Table 1 for an overview).
http://dx.medra.org/10.14614/CHEMOLEUK.2.29
Table 1.
leukaemia
Biomarker
LKBI-AMPK
AMPK
ULKI
P53
PTEN
Atg7
HIF
mTOR kinase
FOXO3A
FUMH
32
Stress biomarker candidates for
Genomic
Protein
+
+
+
+
+
+
+
+
+
+
A possible candidate-biomarker is the serine threonine kinase
ULK1, which is a key initiator of autophagy. Its activation is
essential for clearance of cellular mitochondria and ribosomes
(Kundu et al., 2008). This kinase is activated under growth
factor deprivation and leads to activation of the glycogen
synthesis kinase 3 (GSK-3). The latter phosphorylates the
acetyltransferase TIP60 which in turn aetylates and activates
ULK1 (Lin et al., 2012). Another crucial kinase which is directly
related to autophagy is the mTOR kinase which, under normal
nutrient concentrations, binds and phosphorylates the ULK1,
therefore suppressing autophagy. Under nutrient deprivation,
the activation of the P53 gene enables activation of the ULK1
and autophagy (Feng et al., 2005). Another possible candidate
biomarker is the Atg7 gene, which is an essential gene for activation of autophagy and further regulation of HSC maintenance
(Kundu et al., 2008; Mortensen et al., 2011; Mortensen & Simon,
2010). It has been shown that deleting this gene in murine HSC
leads to death as a result of an accumulation of mitochondria
and ROS, increased proliferation and DNA damage (Mortensen
et al., 2011; Mortensen & Simon, 2010). FOXO3A can also be a
possible biomarker candidate. It has been found to have a critical
role in autophagy induced in mice in a cytokine-free environment
(Warr et al., 2013a).
Several researchers have reported a tumour suppressor role of
autophagy. More specifically, it has been shown that activation
of the AMPK pathway has a suppressor role in AML. AMPK is
a protein kinase which regulates protein and energy homeostasis
at an intracellular level via autophagic recycling of intracellular
components. Practically, AMPK acts as a metabolic sensor
of alteration of the intracellular lipid composition and restores
energy by maintaining the balance ATP vs AMP, through the
LKBI-AMPK activation. LKBI-AMPK is a tumour suppressor
in AML (Green et al., 2010). Activation of the P53/PTEN genes
has been shown to have a tumour suppressive role as these allow
initiation of autophagy via inhibition of the activity of the mTOR
kinase (Feng et al., 2005).
2.3.2
(Non-autophagic) Stress biomarker candidates
A biomarker related to oxidative stress is the Hypoxia Induced
Factor, HIF, which is the central regulator of oxygen homeostasis.
More specifically, HIF1-a regulator is overexpressed in many cancer types and HIF proteins mediate cell adaptation to hypoxia
(Birner et al., 2000; Talks et al., 2000, Warr & Passagué, 2013b;
Zhong et al., 1999).
A possible cell biomarker related to starvation stress is the
metabolic enzyme fumarate hydratase (FUMH) which converts fumarate to malate. This enzyme highly controls the intracellular levels of fumarate, with silencing of the expression of
FUMH leading to fumarate intracellular accumulation (Ratcliffe,
2007).
http://www.um.edu.mt/healthsciences/mjhs
Key environmental stress biomarker candidates for the optimisation of chemotherapy treatment of
leukaemia
3
Conclusion
The impact of environmental stresses such as oxidative and
starvation stress on the leukaemic cell evolution i.e. growth
and/or resistance to drugs, has been discussed in depth. It is clear
that the effect of these factors on the cancer evolution should be
taken into consideration for the accurate prediction of optimal
chemotherapy protocols for specific individuals. Furthermore,
several cellular components that are related to the cell response
to the stress have been analysed. Quantitative information on
these key biomarkers could serve as an appropriate input for
the construction of more detailed predictive models for the in
silico description of the leukaemic evolution. More specifically,
from a detection/quantification point of view and as a first step,
application of techniques such as next generation sequencing
(NGS) or deep sequencing will enable the selection of biomarker
candidates that could be linked to in vitro drug resistance.
These data will enable an accurate model development. As
a second step, screening of patients’ genetic profile through
NGS in BM biopsies, will enable a cross-link of in vivo with
in vitro biomarkers. Consequently, the developed in silico tool
will indicate appropriate and different chemotherapy treatment
options in a closely personalised manner.
Overall, quantification of appropriate (intra-) cellular biomarkers that are related to the leukaemic in vitro kinetics can enable
the combination of macroscopic kinetics with microscopic information, leading to the construction of more detailed models. The
(micro) cellular information that these models account for will
allow a much more accurate prediction of the cancer response,
eventually leading to delivery of personalised chemotherapy
protocols for the treatment of patients with leukaemia.
This review article was compiled as part of the invited
seminar by Dr E.G. Velliou titled ‘A framework for the personalisation/optimisation of the treatment of Acute Myeloid
Leukaemia (AML)’, which was held at the Faculty of Health
Sciences, University of Malta on 4 April 2014.
4
Funding
This work is supported by ERC-BioBlood (no. 340719), ERCMobile Project (no. 226462), the EU 7th Framework Programme [MULTIMOD Project FP7/2007-2013, no. 238013] and
the Richard Thomas Leukaemia Research Fund. Ruth Misener
is further thankful for a Royal Academy of Engineering Research
Fellowship.
5
Conflicts of Interest
The authors report no conflicts of interest.
References
Adbal Dayem, A., Choi, H.-Y., Kim, J.-H. & Cho, S.-G. (2010)
Role of oxidative stress in stem, cancer, and cancer stem
cells. Cancers, 2, pp. 859-884.
American Cancer Society (2013) Available from: www.cancer.org
[Accessed 24th July 2014].
Banerji, V. & Gibson, S.B. (2012) Targeting metabolism and autophagy in the context of haematologic malignancies. International Journal of Cell Biology, Volume 2012, pp. 1-9.
Beutler, E., Coller, B.S., Lichtman, M.A., Kipps, T.J. & Seligsohn, U. (Eds) (2001) Williams Hematology. New York:
McGraw-Hill.
Birner, P., Schind, M., Obermair, A., Plank, C., Breitenecker,
G. & Ober-huber, G. (2000) Overexpression of hypoxiainducible factor 1a is a marker for an unfavorable prognosis
in early-stage invasive cervical cancer. Cancer Research, 60,
http://dx.medra.org/10.14614/CHEMOLEUK.2.29
33
pp. 4693-4696.
Bonnet, D. & Dick, J. (1997) Human acute myeloid leukemia is
organized as a hierarchy that originates from a primitive
hematopoietic cell. Nature Medicine, 3(7), pp. 730-737.
Cancer
Research
U.K.
(2014)
Available
from:
http://www.cancerresearchuk.org.
[Accessed 11th
June 2014].
Choi, A.M.K., Ryter, S.W. & Levine, B. (2013) Autophagy in
human health and disease. The New England Journal of
Medicine, 368, pp. 651-552.
Degenhardt, K., Mathew, R., Beaudoin, B., Bray, K., Anderson,
D., Chen, G., Mukherjee, C., Shi, Y., Gelinas, C., Fan, Y.,
Nelson, D.A. & White, E. (2006) Autophagy promotes tumour cell survival and restricts necrosis, inflammation and
tumori-genesis. Cancer Cell, 10(1), pp. 51-54.
Fecteau, J.-F., Messmer, D., Zhang, S., Cui, B., Chen, L. & Kipps,
T.J. (2013) Impact of oxygen concentration on growth of
mesenchymal stromal cells from the marrow of patients with
chronic lymphocytic leukemia. Blood, 121, pp. 971-974.
Feng, Z., Zhang, H., Levine, A.J. & Jin, S. (2005) The coordinate regulation of the p53 and mTOR pathways in cells.
Proceedings of the National Academy of Science (PNAS),
102(23), pp. 8204-8209.
Fruehauf, J.-P. & Meyskens, F.-L. Jr. (2007) Reactive oxygen
species: a breath of life or death? Clinical Cancer Research,
13, pp. 89-794.
Fuentes-Garí, M., Misener, R., García-Münzer, D.G., Velliou,
E., Panoskaltsis, N., Pistikopoulos, E.N. & Mantalaris,
A. (2014) Development and experimental validation of
cyclin-based population balance model of the cell cycle in
leukaemia cell lines. Journal of Tissue Engineering and
Regenerative Medicine, 8, pp. 489.
García-Münzer, D.G., Kostoglou, M., Georgiadis, M., Pistikopoulos, E.N. & Mantalaris A. (2013) Developing a cyclin
blueprint as a tool for mapping the cell cycle in GS-NS0.
Biochemical Engineering Journal, 81, pp. 97-107.
García-Münzer, D.G., Kostoglou, M., Georgiadis, M.C., Pistikopoulos, E.N. & Mantalaris, A. (2014) A cyclin distributed cell cycle model in GS-NS0. Computer Aided
Chemical Engineering, 33, pp. 19-24.
Giuntoli, S., Tanturli, M., Gesualdo, F.D., Barbetti, V., Rovida,
E. & Sbarba, P.D. (2011) Glucose availability in hypoxia
regulates the selection of chronic myeloid leukemia progenitor subsets with different resistance to imatinib-mesylate.
Haematologica, 96(2), pp. 204-212.
Green, A.S., Chapuis, N., Maciel, T.T., Willems, L., Lambert,
M., Arnoult, C., Boyer, O., Bardet, V., Park, S., Foretz,
M., Viollet, B., Ifrah, N., Dreyfus, F., Hermine, O., Moura,
I.C., Lacombe, C., Mayeux, P., Bouscary, D. & Tamburini,
J. (2010) The LKB1/AMPK signaling pathway has tumour
suppressor activity in acute myeloid leukemia through the
repression of mTOR-dependent oncogenic mRNA translation. Blood, 116, pp. 4262-4273.
Herst, P.M., Howman, R.A., Neeson, P.J., Berridge, M.V.&
Ritchie D.S. (2011) The level of glycolytic metabolism in
acute myeloid leukemia blasts at diagnosis is prognostic for
clinical outcome. Journal of Leukocyte Biology, 89, pp. 5155.
Hippest, M.M., O’Toole, P.S. & Thorburn, A. (2006) Autophagy
in cancer: good, bad or both? Cancer Research, 66, pp.
9349-9351.
Jin, S. & White, E. (2007) Role of autophagy in cancer: management of metabolic stress. Autophagy, 3(1), pp. 28-31.
Kipps, T.J. (2001) Classification of malignant lymphoid disorders.
In E. Beutler, B.S. Coller, M.A. Lichtman, T.J. Kipps & U.
Seligsohn (Eds) Williams Hematology. New York: McGrawHill.
Kongara, S. & Karantza, V. (2012) The interplay between au-
http://www.um.edu.mt/healthsciences/mjhs
Key environmental stress biomarker candidates for the optimisation of chemotherapy treatment of
leukaemia
tophagy and ROS in tumorigenesis. Frontiers in Oncology,
2(171), pp. 1-13.
Kundu, M., Lindsten, T., Yang, C.-Y., Wu, J., Zhao, F., Zhang,
J., Selak, M.A., Ney, P.A. & Thompson, C.B. (2008) Ulk1
plays a critical role in the autophagic clearance of mitochondria and ribosomes during reticulocyte maturation. Blood,
112, pp. 1493-1502.
Levine, B. & Kroemer, G. (2008) Autophagy in the pathogenesis
of disease. Cell, 132(1), pp. 27-42.
Lichtman, M.A. (2001) Classification and clinical manifestations
of the clonal myeloid disorders. In E. Beutler, B.S. Coller,
M.A. Lichtman, T.J. Kipps & Seligsohn, U. (Eds) Williams
Hematology. New York: McGraw-Hill.
Lin, S-Y., Li, T.Y., Liu, Q., Zhang, C., Li, X., Chen, Y., Zhang,
S-M., Lian, G., Liu, Q., Ruan, K., Wang, Z., Zhang, C.S.,
Chien, K-Y., Wu, J., Li, Q., Han, J. & Lin, S.C. (2012)
GSK3-TIP60-ULK1 Signaling pathway links growth factor
deprivation to autophagy. Science, pp. 336-477.
Liu, R., Li, Z., Bai, S., Zhang, H., Tang, M., Lei, Y., Chen, L.,
Liang, S., Zhao, Y.-I., Wei, Y. & Huang, C. (2009) Mechanism of cancer cell adaptation to metabolic stress: proteomics identification of a novel thyroid hormone-mediated
gastric carcinogenic signaling pathway. Molecular and Cellular Proteomics, 8, pp. 70-85.
Lodi, A., Tiziani, S., Khanim, F.L., Drayson, M.T., Gunther,
U.L., Bunce, C.M. & Viant, M.R. (2011) Hypoxia triggers major metabolic changes in AML cells without altering
indomethacin-induced TCA cycle deregulation. American
Chemical Society (ACS ) Chemical Biology, 6, pp. 169-175.
Lowenberg, B., Downing, J. & Burnett, A. (1999) Acute Myeloid
Leukemia. New England Journal of Medicine, 341(14), pp.
1051-1062.
Lozy, F. & Karantza, V. (2012) Autophagy and cancer cell
metabolism. Seminars in Cell and Developmental Biology,
23, pp. 395-401.
Lum, J., Bauer, D.E., Kong, M., Harris, M.H., Li, C., Lindsten, T.
& Thompson, C.B. (2005) Growth factor regulation of autophagy and cell survival in the absence of apoptosis. Cell,
120, pp. 237-248.
Lyu, B.N., Ismailov, S.B., Ismailov, B. & Lyu, M.B. (2008) Mitochondrial concept of leukemogenesis: key role of oxygenperoxide effects. Theoretical Biology and Medical Modelling, 5(23), pp. 1-11.
Moore, M.N., Allen, J.I. & Somerfield, P.J. (2006) Autophagy:
role in surviving environmental stress. Marine Environmental Research, 62, S420-S425.
Mortensen, M. & Simon, A.K. (2010) Nonredundant role of Atg7
in mitochondrial clearance during erythroid development.
Autophagy, 6, pp. 423-425.
Mortensen, M., Soilleux, E.J., Djordjevic, G., Tripp, R., Lutteropp, M., Sadighi-Akha, E., Stranks, A.J., Glanville, J.,
Knight, S., Jacobsen, S.-E.W., Kamil, R., Kranc, K.R. &
Simon, A.K. (2011) The autophagy protein Atg7 is essential for hematopoietic stem cell maintenance. The Journal
of Experimental Medicine, 208(3), pp. 455-467.
Mortensen, M., Watchon A.S. & Simon, A.K. (2011) Lack of
autophagy in the hematopoietic system leads to loss of
hematopoietic stem cell function and dysregulated myeloid
proliferation. Autophagy, 7(9), pp. 1069-1070.
Mortera-Blanco T., Mantalaris, A., Bismarck, A. & Panoskaltsis,
N. (2010) Development of a three-dimensional biomimicry
of human acute myeloid leukemia ex vivo. Biomaterials, 31,
pp. 2243-2251.
Panoskaltsis, N., Reid, C.D.L. & Knight, S.C. (2003) Quantification and cytokine production of circulating lymphoid
and myeloid cells in acute myelogenous leukemia (AML).
Leukemia, 17, pp. 716-730.
Passegué, E., Jamieson, C.H.M., Ailles, L.E. & Weissman, I.L.
http://dx.medra.org/10.14614/CHEMOLEUK.2.29
34
(2003) Normal and leukemic hematopoiesis: are leukemias
a stem cell disorder or a reacquisition of stem cell characteristics? Proceedings of the National Academy of Science
(PNAS), 100, 11842-11849.
Pefani, E., Panoskaltsis, N., Mantalaris, A., Georgiadis, M.C.
& Pistikopoulos, E.N. (2013) Design of optimal patientspecific chemotherapy protocols for the treatment of acute
myeloid leukemia (AML). Computers and Chemical Engineering, 57, pp. 187-195.
Pefani, E., Panoskaltsis, N., Mantalaris, A., Georgiadis, M.C.
& Pistikopoulos, E.N. (2014) Chemotherapy drug scheduling for the induction treatment of patients diagnosed with
Acute Myeloid Leukemia. Transactions of Biomedical Engineering Journal, 61(7), pp. 2059-2056.
Pui, C.H. (2009) Acute Lymphoblastic Leukaemia: introduction.
Seminars in Hematology, 46(1), pp. 1-2.
Pui, C.H. & Evans, W.E. (2006) Treatment of acute lymphoblastic
leukemia. The New England Journal of Medicine, 354, pp.
166-178.
Ratcliffe, P. J. (2007) Fumarate hydratase deficiency and cancer:
activation of hypoxia signaling? Cancer Cell, 11, pp. 303305.
Scherz-Shouval, E., Shvets, E., Fass, E., Shorer, H., Gil, L. &
Elazar, Z. (2008) Reactive oxygen species are essential for
autophagy and specifically regulate the activity of Atg4.
The EMBO Journal, 26, pp. 1749-2760.
Talks, K. L., Turley, H., Gatter, K. C., Maxwell, P. H., Pugh, C.
W., Ratcliffe, P. J. & Harris, A. L. (2000) The expression
and distribution of the hypoxia-inducible factors HIF-1a
and HIF-2a in normal human tissues, cancers, and tumourassociated macrophages. American Journal of Pathology,
157, pp. 411- 421.
Velliou, E., Fuentes-Gar’, M., Misener, R., Pefani, E., Rende,
M., Panoskaltsis, N., Pistikopoulos, E. N. & Mantalaris A.
(2014a) A framework for the design, modeling and optimisation of biomedical systems. Computer Aided Chemical
Engineering, 34, pp. 225-236.
Velliou, E., Brito Dos Santos, S., Papathanasiou, M., FuentesGar’ M., Misener R., Panoskaltsis, N., Mantalaris, A. &
Pistikopoulos, E.N. (2014b) Evolution of an AML model
system under oxidative and starvation stress: a comparison
between two and three dimensional cultures. Journal of
Tissue Engineering and Regenerative Medicine, 8, pp. 383.
Wallington-Beddoe, C.T., Hewson, J., Bradstock, K.F. & Bendall, L.J. (2011) FTY720 produces caspase-independent cell
death of acute lymphoblastic leukaemia cells. Autophagy,
7(7), pp. 707-715.
Warr, M.R., Binnewies, M., Flach, J., Reynaud, D., Garg, T.,
Malhorta, R., Debnath, J. & Passegu’, E. (2013) FOXO3A
directs a protective autophagy program in hematopoietic
stem cells. Nature, 494, pp. 323-327.
Warr, M.R. & Passegu’, E. (2013) Metabolic makeover for HSCs.
Cell Stem Cell, 12, pp. 1-3.
Wilkinson, S.T., Tome, M.E. & Briehl, M.M. (2012) Mitochondrial adaptations to oxidative stress confer resistance to
apoptosis in lymphoma cells. International Journal of
Molecular Sciences, 13, pp. 10212-10228.
Zhong, H., Marzo, A. M., Laughner, E., Lim, M., Hilton, D. A.,
Zagzag, D., Buechler, P., Isaacs, W. B., Semenza, G. L. &
Simons, J. W. (1999) Overexpression of hypoxia-inducible
factor 1 alpha in common human cancers and their metastases. Cancer Research, 59, pp. 5830’5835.
Zhou, F.-L., Zhang, W.-G., Wei, Y.-C., Meng, S., Bai, G.-G., BaiWang, B.-Y., Yang, H.-Y. , Tian, W., Meng, X., Zhang,
H. & Chen, S.-P. (2010) Involvement of oxidative stress in
the relapse of Acute Myeloid Leukemia. The Journal of
Biological Chemistry, 285, pp. 15010-15015.
http://www.um.edu.mt/healthsciences/mjhs
Malta Journal of Health Sciences
http://www.um.edu.mt/healthsciences/mjhs
DOI: http://dx.medra.org/10.14614/BURNPOD.2.38
Research paper
DEGREE OF BURNOUT AMONG MALTESE
WORKING IN TWO DIFFERENT SETTINGS
PODIATRISTS
Trista Agius1 , Cynthia Formosa2
1
Podiatry Clinic (Primary Health Care), Mosta Health Centre, Mosta, Malta
2
Department of Podiatry, Faculty of Health Sciences, University of Malta, Msida, Malta
Abstract. The issue of burnout is receiving increasing research attention. However, most of the literature on burnout has
focused on the nursing, physiotherapy and occupational therapy
professions, with very few studies within the podiatric profession
exploring this concept. The purpose of this paper is to compare
the degree of burnout among Maltese podiatrists working in two
different settings, namely the public health service and private
practice. A quantitative non-experimental study, involving a
convenience sample, was conducted. The Copenhagen Burnout
Inventory (CBI) was used to evaluate levels of burnout in the two
participant groups. No statistical difference was found between
the overall mean burnout scores for Maltese podiatrists employed
in the public health service and those working in private practice
(p = 0.067, mean = 48.89 vs 31.84). Although the level of
burnout did not differ between podiatrists working within the
two different settings, podiatrists working in the public sector
in general reported higher mean scores for each subscale of the
CBI than podiatrists working in private practice. More research
is necessary to build a better understanding of burnout among
podiatrists in parallel with other health care professionals, in
order to help prevent or alleviate this phenomenon.
Keywords: occupational burnout, podiatrists, public health service, private practice, performance, job satisfaction
1
Introduction
The literature suggests that burnout is a serious problem which
needs to be addressed appropriately (Akroyd, Caison & Adams,
2002; Akgun, Al-Assaf & Bakan, 2008). Burnout emerged as
an important concept in the 1970s and continues to do so today (Schaufeli, Leiter & Maslach, 2008). Burnout is regarded as
a multidimensional syndrome which comprises three components,
namely emotional exhaustion, depersonalisation and reduced personal accomplishment (Brenninkmeijer & Van Yperen, 2003).
Burnout syndrome can have deliberate implications for health
care professionals, their patients and the organisation (Lloyd &
King, 2004). For this reason, research on burnout syndrome is
Correspondence to: Cynthia Formosa
(cynthia.formosa@um.edu.mt)
Received: 08.02.14 - Revised: 16.10.14 - Accepted: 16.10.14 Published: 17.12.14
c 2014 The authors
increasing, particularly in the health service professions. There
has been a continuous search among researchers to develop improved burnout measures to study this concept. These include the
Maslach Burnout Inventory (Schaufeli et al., 1996), the Oldenburg
Burnout Inventory (Demerouti et al., 2001) and the more recent
Copenhagen Burnout Inventory (CBI) (Kristensen et al., 2005).
All these instruments embrace the assumption that the burnout
concept comprises all the three components discussed above.
Burnout is a distinct kind of occupational stress which generally
occurs among health care professionals, mainly due to their continual exposure to patients’ problems, although the point at which
burnout begins is unclear (Mandy & Tinley, 2004). This in turn
results in emotionally drained and burnt out professionals (Akroyd
et al., 2002). Burnout is known to result in social and medical
problems in the professional, such as dissatisfaction, frustration,
anxiety, detachment, psychological fatigue, headaches, sleep disorders, intention to quit the job, depression and substance abuse
(Akgun et al., 2008; Schaufeli & Enzmann, 1998). Angerer (2003)
reported that this concept, which can lead to cynicism in the individual’s work performance, is correlated with absenteeism, job
turnover and low productivity, reduced job satisfaction and decreased commitment to the job. Failing to identify burnout among
workers will inevitably lead to considerable financial, social and
psychological costs (Maslach & Leiter, 1997; Glasberg, Norber &
Soderberg, 2007).
In Malta, the podiatric profession is still relatively new when
compared to other local health care professions. Podiatry exists
as a clinically independent profession involving the diagnosis and
treatment of foot problems. The profession has grown considerably since the patient population is also growing, with 52% to 90%
of older people complaining of a foot problem. In Malta, podiatry
is providing its services to over 60,000 people annually through
the public health service only (The Association of Podiatrists of
Malta, 2007). A podiatrist is a qualified health professional who
specialises in the prevention, diagnosis, treatment and rehabilitation of disorders of the foot and ankle. Underlying lower limb
structures, such as the knee and the upper third of the lower
limb, may also be examined by a podiatrist since these structures
might be related to a foot or ankle problem. The complex mechanics of the feet (52 bones, a network of muscles, joints and
ligaments) and a wide range of foot problems (over 200 identified
conditions) demand a separate and autonomous profession which
provides high levels of training and professional expertise in the
field of podiatry (The Association of Podiatrists of Malta, 2007).
As a result, the podiatrist’s scope of practice is diverse and has
expanded to include curative and preventive strategies in paediatrics, diabetes, sports injuries, structural and biomechanical
problems, geriatrics, vascular and podiatric surgery (The Associ-
Degree of burnout among Maltese podiatrists working in two different settings
ation of Podiatrists of Malta, 2012). There are currently approximately 66 state-registered podiatrists within the Council for the
Professions Complementary to Medicine of Malta. These professionals work in a variety of settings including the public health
service, private practice, education and research, or as a part of a
multi-disciplinary team operating in a specific specialised clinic.
Most of the literature on burnout has addressed the nursing, physiotherapy and occupational therapy professions (Burke,
Koyuncu & Fiksenbaum, 2010). A small number of studies has
been conducted within the podiatric profession. It has been argued that the relationship between podiatrists and their patients
differs from that in other professions due to the fact that podiatrists are often required to spend extensive time with their patients on a one-to-one basis rather than working in teams with
other health care professionals, possibly resulting in higher levels
of work stress and burnout (Mandy, Lucas & McInnes, 2003).
A comparative research study on burnout among Australian
and British podiatrists was carried out by Mandy and Tinley in
2004. Results suggest that levels of burnout among podiatrists
were higher than those indicated by published normative data for
all health care professionals. These results are of great concern to
the podiatric profession and have prompted the present investigation that addresses this concept in the Maltese culture. The latter
identifies the Maltese population as unique, distinguishing it from
its European neighbours. The Maltese culture is broadly Mediterranean, but it is at the same time very distinctive: it has its own
unique blend of historical and economic traditions which in turn
have influenced the values, motivations, expectations and practices which characterise the Maltese people (Baldacchino, 2000).
The aim of this study was to compare levels of burnout in Maltese podiatrists working in two different settings, namely the private or the public sector only. The nature of work undertaken by
each group was fundamentally the same; it was the environment
that the profession was practised in which differed between the
two groups. The null hypothesis which was formulated for this
study suggested ‘no difference in the levels of burnout between
Maltese podiatrists working in the public health service and those
working in private practice only’.
2
Methods
A quantitative non-experimental study design was employed.
Ethical approval was sought and later granted by the University
of Malta Research Ethics Committee. Convenience sampling was
adopted. The Register of the Council for the Professions Complementary to Medicine was used to identify potential participants.
An information letter was sent to all Maltese podiatrists working either in private practice or within the public health service
only. A copy of the Copenhagen Burnout Inventory (CBI), together with a data sheet which requested details on the nature
of employment, the length of time since qualification, age, gender, marital status and the number of patients seen each week,
was also sent, together with a stamped, self-addressed envelope.
Anonymity and confidentiality of information provided was ensured. All 18 podiatrists working in Malta on a full-time basis
either in private practice or within the public health service participated in the study (response rate = 100%).
2.1
The Copenhagen Burnout Inventory
(CBI)
The CBI is a questionnaire that measures the degree of psychological fatigue experienced in three sub-dimensions of burnout:
personal, work-related and patient-related. The personal burnout
scale is generic, in that it can be answered by any individual.
The work-related burnout scale assumes that the person undertaking the questionnaire has some kind of paid employment, while
the patient-related burnout scale assumes that the individual’s
http://dx.medra.org/10.14614/BURNPOD.2.38
36
work involves patients (Kristensen et al., 2005). This inventory
is unique since its scales were developed in such a way that they
should be able to indicate whether individual feelings of burnout
are due to personal or work-related factors (Kristensen et al.,
2005). Translations of the CBI into eight languages are available, allowing its use in many studies worldwide. Results support
the reliability and validity of the tool, with high internal consistencies ranging from 0.85 to 0.87 (Borritz et al., 2006). The CBI
questionnaire consists of 19 items with five response categories
for each question: Always, Often, Sometimes, Seldom, Never/Almost, Never. For some of the questions, the response categories
are adjusted as follows: To a very high degree, To a high degree,
Somewhat, To a low degree, To a very low degree. Participants
are required to choose only one answer for every question. The
response categories are re-scaled to a 0-100 metric (0 = Never/Almost Never/To a very low degree, 25 = Seldom/To a low degree, 50 = Sometimes/Somewhat, 75 = Often/To a high degree,
100 = Always/To a very high degree. If less than three questions are answered from scales 1 and 3, or less than four questions
are answered from scale 2, the respondent is classified as a nonresponder. A mean rating score greater than 50 indicates a high
degree of burnout, whilst a mean rating score smaller than 50
implies a lower degree of burnout. The CBI is relatively easy to
complete, requiring approximately 15 minutes to be filled in. It is
a public domain questionnaire, unlike other measures of burnout
which are protected by copyright and distributed by a commercial
publisher, thus making it more accessible to researchers. Permission was obtained from the authors of the CBI (Kristensen et al.,
2005) to employ the original English version of the CBI in the
present study. Evidence confirming the reliability and validity of
the instrument enhanced the study’s methodological rigour.
3
Results
Twelve male and six female podiatrists working on a full-time
basis either in private practice or in the public health service participated in this study. Participants were aged between 25 and
45 years, with 10 participants aged between 25 and 35 years and
eight participants aged between 36 and 45 years. Sixty-one percent of the sample was married. Seventy-two percent (N = 13) of
the participants worked within the public health service and 27%
(N = 5) worked in private practice. Ninety-five percent of the
respondents reported seeing approximately 31 to 40 patients per
week.
3.1
CBI Scores
The Kolmogorov-Smirnov test showed collected data to be normally distributed. The one-way analysis of variance (ANOVA)
test was used to confirm or reject the null hypothesis which was
formulated for this study, namely that there was no difference in
burnout levels between podiatrists working in the two different
settings. One-way ANOVA is a parametric test which compares
the means of several independent samples to make assumptions
on the population means (Bowling, 2002). When the p-value was
found to be less than the 0.05 level of significance, it was deduced
that the mean rating scores for the CBI differed significantly, thus
leading to rejection of the null hypothesis.
No statistical difference was found between the overall mean
burnout scores of podiatrists working in the public health service
and private practice (p = 0.067). Moreover, the overall mean
burnout score was less than 50 for each group, implying a low degree of burnout being present among Maltese state-registered podiatrists (mean score for public sector podiatrists = 48.87; mean
score for private practice podiatrists = 31.84). Individual mean
burnout scores were higher for podiatrists working within the public sector when compared to podiatrists working in the private
sector for nearly all items of the three subscales of the CBI, as
illustrated in Tables 1, 2 and 3.
http://www.um.edu.mt/healthsciences/mjhs
Degree of burnout among Maltese podiatrists working in two different settings
37
Table 1. CBI Scale 1 - Personal Burnout: means and standard deviations for the participants (N = 18).
Mean
Std. Deviation
p-value
How often do you feel tired?
Public sector
61.54
24.185
0.769
Private sector
65.00
13.693
How often are you physically exhausted?
Public sector
55.77
27.298
0.687
Private sector
50.00
25.000
How often are you emotionally exhausted?
Public sector
57.69
25.789
0.104
Private sector
35.00
22.361
How often do you think ‘I can’t take it any more’ ?
Public sector
48.08
29.689
0.039
Private sector
15.00
22.361
How often do you feel worn out?
Public sector
57.69
23.683
0.083
Private sector
35.00
22.361
How often do you feel weak and susceptible to illness?
Public sector
50.00
22.822
0.014
Private sector
20.00
11.180
Table 2. CBI Scale 2 -Work Burnout: means and standard deviations for the participants (N = 18).
Mean
Std. Deviation
p-value
Is your work emotionally exhausting?
Public sector
51.92
31.394
0.190
Private sector
30.00
27.386
Do you feel burnt out because of your work?
Public sector
57.69
23.683
0.104
Private sector
35.00
28.504
Does your work frustrate you?
Public sector
32.69
27.735
0.862
Private sector
30.00
32.596
Do you feel worn out at the end of the working
day?
Public sector
59.62
26.098
0.250
Private sector
45.00
11.180
Are you exhausted in the morning at the thought
of another day at work?
Public sector
40.38
29.823
0.057
Private sector
10.00
22.361
Do you feel that every working hour is tiring for
you?
Public sector
40.38
21.743
0.074
Private sector
15.00
33.541
Do you have enough energy for family and friends
during leisure?
Public sector
67.31
27.735
0.877
Private sector
65.00
28.504
4
Discussion
The aim of this study was to compare the degree of burnout among
Maltese podiatrists working in two different settings (private versus public sector). It is important to note that this study captured
the views of all Maltese podiatrists who were working in either the
private or public sector only at the time of the investigation (100%
response rate).
Identification of burnout using the CBI requires participants to
obtain high scores on all three subscales, namely personal burnout,
work-related burnout and patient-related burnout (Borritz et al.,
2006). No statistical difference between mean burnout scores of
podiatrists working in the public health service and private prac-
http://dx.medra.org/10.14614/BURNPOD.2.38
tice was identified. Moreover, the mean burnout scores for each
subscale of the CBI were less than 50 for each group, implying
a low degree of burnout among Maltese state-registered podiatrists. Previous study results on burnout among podiatrists in
other populations are not congruent with the results of this study.
Podiatrists in the United Kingdom and Australia were reported
to exhibit high levels of emotional exhaustion and depersonalisation, as well as low levels of personal accomplishment when using
the Maslach Burnout Inventory and the Work Stress Inventory
to measure burnout levels (Mandy, 2000; Mandy & Tinley, 2002,
2004). However, this is the first study to have explored burnout
within the Maltese context. Furthermore, the tool used to measure burnout in this specific population of podiatrists is differ-
http://www.um.edu.mt/healthsciences/mjhs
Degree of burnout among Maltese podiatrists working in two different settings
ent from the ones employed in the studies mentioned above. It
could be argued that the limitations of this study include that
all data were collected using a self-report questionnaire, raising
the possibility of response set tendencies. Furthermore, the data
were collected at one point in time, making it difficult to determine causality. Participants’ personality and past or recent traumas/losses were not taken into consideration.
When comparing the overall mean burnout scores between the
two groups, podiatrists working in the public sector demonstrated
a higher score than those working in the private sector. Research
carried out among various health care professionals has led to
the conclusion that health care workers within the public sector
exhibit a higher degree of burnout when compared to those in
private practice (Mrayyan, 2005; Pillay, 2009; Pinto, 2009). Various reasons have been given for this outcome, including that the
private sector is less afflicted by rules, regulations and restrictive
work, and that motivation in this sector is highly based on profits
(Macklin, Smith & Dollard, 2006; Cherniss, 1995).
Consideration of the Personal Burnout CBI subscale demonstrates that podiatrists working within the public sector reported
feeling more physically and emotionally exhausted and more worn
out than podiatrists working privately. On the Work Burnout
subscale, podiatrists employed within the public sector reported
being more emotionally exhausted at work, feeling more burnt out
as a result of their work, feeling more frustrated with their work,
feeling more worn out at the end of a working day and also feeling
more exhausted at the thought of another day at work when compared to podiatrists working in the private sector. However, it is
interesting to note that despite these negative responses, podiatrists working in the public sector reported more energy for family
and friends after work than those working in the private sector. As
for the Patient Burnout scale, podiatrists working within the public sector reported that, when working with patients, they found
it harder to work, were more frustrated, felt more drained and
felt that they gave more than they got back when compared to
podiatrists working within the private sector.
The patient-clinician relationship can differ across the public
and private sectors. A number of common themes have been identified across studies, explaining why podiatrists working within
the public sector tend to be more burnt out than podiatrists working in the private sector. These include lack of professional status
among colleagues and patients, as well as labeling by patients as
38
being unproductive, inefficient, distant and poorly motivated (Ferrinho et al., 2004). Long waiting lists are attributed to health care
professionals working inside the public sector, leading them to being viewed as incompetent and disrespectful (Forrest et al., 2002).
Over-demanding patients, shortage of staff and professional isolation have also been reported to result in professional burnout
(Mandy & Tinley, 2004). Moreover, patients attending private
services considered their practitioners to be more understanding
than practitioners working in the public sector and reported that
clinicians had more time available to spend with them (Bjorngaard
et al., 2008). Feedback from patients is important for any health
care professional to feel appreciated (Mrayyan, 2005). Maslach
and Leiter (1997) argue that feedback from patients attending
public health care services is either non-existent or almost always
negative. Patients attending the public sector do not pay directly
for services rendered and thus are more likely to neglect and ignore advice given to them by their clinicians. This in turn leaves
health care professionals feeling frustrated, tired and exhausted.
Further research investigating differences in burnout between
different podiatric clinical environments in this specific population
is warranted if a better service is to be offered to patients attending podiatric services. A better understanding of the burnout
concept could be developed through the use of longitudinal research designs that also address off-job experiences in the context
of burnout research, particularly addressing indicators of psychological well-being as predictors of work outcomes, in addition to
using the three CBI subscales (Sonnentag, 2005). Such studies
should enable a better understanding of burnout and why it happens. Results could also provide information on how to cope with
it, prevent it or combat it (Schaufeli et al., 2008). Prevention and
management of burnout is of utmost importance since it is not
just a personal problem but it could also have significant implications for organisations’ performance. More motivated podiatrists
have a better quality of life, which in turn translates into improved
patient care (Mandy & Tinley, 2004).
5
Conclusion
The results of this study have shown no difference in burnout
between Maltese podiatrists working in the public and private
sectors. However, the podiatrists working in the public sector
Table 3. CBI Scale 3 - Patient Burnout: means and standard deviations for the participants (N = 18).
Mean
Std. Deviation
p-value
Do you find it hard to work with patients?
Public sector
34.62
26.098
0.134
Private sector
15.00
13.693
Do you find it frustrating to work with patients?
Public sector
32.69
21.371
0.044
Private sector
10.00
13.693
Does it drain your energy to work with patients?
Public sector
48.08
29.689
0.256
Private sector
30.00
27.386
Do you feel that you give more than you get back
when you work with patients?
Public sector
61.54
24.185
0.082
Private sector
40.00
13.693
Are you tired of working with patients?
Public sector
30.77
25.318
0.241
Private sector
15.00
22.361
Do you sometimes wonder how long you will be
able to work with patients?
Public sector
40.38
33.132
0.794
Private sector
45.00
32.596
obtained higher mean burnout scores for most items of the CBI
http://dx.medra.org/10.14614/BURNPOD.2.38
http://www.um.edu.mt/healthsciences/mjhs
Degree of burnout among Maltese podiatrists working in two different settings
subscales when compared to the practitioners working privately.
More research is warranted in this area of study in Malta to contribute towards a better understanding of burnout among podiatrists in parallel with other health care professionals, in order
to help prevent or alleviate this phenomenon. Furthermore, results of this study encourage further research with the purpose of
establishing the CBI’s validity and reliability in the local context.
6
Acknowledgements
The authors would like to thank all podiatrists who agreed to
participate in this study. The authors are also grateful to Marianne Borritz who gave them permission to use the English version
of the CBI.
7
Funding
This research has received no specific grant from any funding
agency in the public, commercial or non-profit sectors.
8
Conflicts of Interest
The authors report no conflicts of interest.
References
Akgun, S., Al-Assaf A.F. & Bakan, C. (2008) Reducing burnout
among hospital professionals: EAPs can help identify
and alleviate the factors that cause burnout and improve
the health and performance of hospital staff. Available
from:
http://www.avitadestek.com/media/img_basin/
joea.pdf. [Accessed 29th July 2013].
Akroyd, D., Caison, A. & Adams, R. (2002) Patterns of burnout
among U.S. radiographers. Radiologic Technology, 73(33),
pp. 215-223.
Angerer, J.M. (2003) Job burnout. Journal of Employment Counseling, 40, pp. 98-107.
Baldacchino, G. (2000) Introducing Social Studies: A Maltese
reader. Malta: PEG Ltd.
Bjorngaard, J.H., Garratt, A., Grave, R.W., Bjertnes, O.A. &
Ruud, T. (2008) Patient experiences with treatment in private practice compared with public mental health services.
Scandinavian Journal of Psychology, 49, pp. 385-392.
Borritz, M., Rugulies, R., Bjorner, J. B., Villadsen, E., Mikkelsen,
O. A. & Kristensen, T. S. (2006) Burnout among employees
in human service work: design and baseline findings of the
PUMA study. Scandinavian Journal of Public Health, 34
(1), pp. 49-58.
Bowling, A. (2002) Research Methods in Health: Investigating
health and health services (2nd ed.). Buckingham: Open
University Press.
Brenninkmeijer, V. & Van Yperen, N. (2003) How to conduct
research on burnout: advantages and disadvantages of a
unidimensional approach in burnout research. Occupational
and Environmental Medicine, 60 (Suppl 1): i16-i20.
Burke, R.J., Koyuncu, M. & Fiksenbaum, L. (2010) Burnout, work
satisfaction and psychological well-being among nurses in
Turkish hospitals. Europe’s Journal of Psychology, 1, pp.
63-81.
Cherniss, C. (1995) Beyond Burnout: Helping teachers, nurses,
therapists and lawyers recover from stress and disillusionment. London: Routledge.
Demerouti, E., Bakker, A.B., Nachreiner, F. & Schaufeli, W.B.
(2001) Job demands resources of burnout. Journal of Applied Psychology, 86, pp. 499-512.
Ferrinho, P., Lerberghe, W.V., Fronteira, I., Hipólito, F. & Biscaia, A. (2004) Dual practice in the health sector: review
of the evidence. Human Resources for Health, 2(14), pp.
http://dx.medra.org/10.14614/BURNPOD.2.38
39
1-17.
Forrest, C.B., Shi, L., Schrader, S.V. & Ng, J. (2002) Managed
care, primary care, and the patient-practitioner relationship. Journal of General Internal Medicine, 17, pp. 270277.
Glasberg, A.L., Norber, A. & Soderberg, A. (2007) Sources of
burnout among health care employees as perceived by managers. Journal of Advanced Nursing, 60(1), pp. 10-19.
Kristensen, T.S., Borritz, M., Villadsen, E. & Christensen, K.B.
(2005) The Copenhagen Burnout Inventory: A new tool for
the assessment of burnout. Work and Stress, 19(3), pp.
192-207.
Lloyd, C. & King, R. (2004) A survey of burnout among Australian mental health occupational therapists and social
workers. Social Psychiatry and Psychiatric Epidemiology,
39, pp. 752-757.
Macklin, D.S., Smith, L.A. & Dollard, M.F. (2006) Public and
private sector work stress: workers’ compensation, levels
of distress and job satisfaction, and the demand-controlsupport model. Australian Journal of Psychology, 58(3),
pp. 130-143.
Mandy, A. (2000) Burnout and work stress in newly qualified podiatrists in the NHS. British Journal of Podiatry, 3(2), pp.
31-34.
Mandy, A., Lucas, K. & McInnes, J. (2003) Psychosocial Approaches to Podiatry; A companion for practice. United
Kingdom: Churchill Livingstone.
Mandy, A. & Tinley, P. (2002) Burnout and occupational stress
in Australian podiatrists. Australasian Journal of Podiatric
Medicine, 36, pp. 101-108.
Mandy, A. & Tinley, P. (2004) Burnout and occupational stress:
comparison between United Kingdom and Australian podiatrists. Journal of the American Podiatric Medical Association, 94(3), pp. 282-291.
Maslach, C. & Leiter, M.P. (1997) The Truth About Burnout,
How Organisations Cause Personal Stress and What To
Do About It. USA: John Wiley & Sons.
Mrayyan, M.T. (2005) Nurse job satisfaction and retention: comparing public to private hospitals in Jordan. Journal of
Nursing Management, 13, pp. 40-50.
Pillay, R. (2009) Work satisfaction of professional nurses in South
Africa: a comparative analysis of the public and private
sectors. Human Resources for Health, 7(15), pp. 1-10.
Pinto, C. (2009) Work stress, satisfaction and burnout in New
Zealand radiologists: comparison of public hospital and private practice in New Zealand. Journal of Medical and Radiation Oncology, 53(2), pp. 194-199.
Schaufeli, W.B. & Enzmann, D. (1998) The Burnout Companion
to Study and Practice: A critical analysis. London: Taylor
& Francis.
Schaufeli, W.B., Leiter M.P. & Maslach C. (2008) Burnout: 35
years of research and practice. Career Development International, 14(3), pp. 204-220.
Schaufeli, W.B., Leiter M.P., Maslach C. & Jackson, S.E. (1996)
Maslach Burnout Inventory General Survey (MBI GS). In
C. Maslach, S.E. Jackson and M.P. Leiter (Eds) Maslach
Burnout Inventory (3rd ed.). Palo Alto, CA: Consulting
Psychologists Press.
Sonnentag, S. (2005) Burnout research: adding an off-work and
day-level perspective. Work and Stress, 19(3), pp. 271-275.
The Association of Podiatrists of Malta (2007) History of Podiatry. Available from http://www.owenwebhosting.com/
apm/HistoryPodiatry.htm 2007. [Accessed 29th July 2012].
The Association of Podiatrists of Malta (2012) The Podiatrist.
Available from podiatry.org.mt/en/home.htm. [Accessed
22nd September 2013].
http://www.um.edu.mt/healthsciences/mjhs
Malta Journal of Health Sciences Online
http://www.um.edu.mt/healthsciences/mjhs
Doi: http://dx.medra.org/10.14614/COMMAUT.2.40
Research paper
THE EFFECTIVENESS OF SOCIAL COMMUNICATION GROUPS
FOR CHILDREN WITH AUTISM SPECTRUM DISORDER IN MAINSTREAM SCHOOLS
Tiziana Baldacchino, Kristina Agius
Department of Communication Therapy, Faculty of Health Sciences, University of Malta, Msida, Malta
Abstract.
The purpose of this study was to evaluate
the effectiveness of a 10-week Social Communication Group
(SCG) programme involving six children diagnosed with Autism
Spectrum Disorder (ASD). The sessions were carried out at
the mainstream school attended by the children. This study
evaluated the effectiveness of the programme in enhancing
specific social communication skills in each participant, as
opposed to previous research on social skills groups where results
were more general. The children’s social communication skills
were rated pre- and post-therapy by the first author, parents
and Learning Support Assistants (LSAs) to allow comparison.
Feedback forms filled in by the LSAs and the first author’s
own observations allowed ongoing evaluation of each child’s
performance throughout sessions. Questionnaires given pre- and
post-therapy to the parents and LSAs were expected to provide
evidence of generalisation of social communication skills at home
and at school. Qualitative and quantitative data obtained from
this small group study indicated that this method of intervention
was effective in enhancing the social communication skills of
children with ASD. Although all the participants showed an
improvement in their overall performance, specific objectives
targeted through structured activities that were motivating and
repetitive elicited a higher percentage improvement. Factors
such as the children’s primary language, pre-therapy status and
their understanding at conversational level seemed to have an
impact on their individual improvement. Generalisation of skills
acquired during therapy was limited since it depended on the
commitment of parents and LSAs in implementing the goals of
the programme at home and at school.
Keywords Autism Spectrum Disorder, social communication, group therapy, mainstream school, case study
1
Introduction
The development of social communication is necessary for the
adequate use of verbal and non-verbal communication for social
purposes, across various contexts and with different communication partners (Prizant & Wetherby, 2005). According to the fifth
Correspondence to: Tiziana Baldacchino (tiziana6@gmail.com)
Received: 04.08.14 - Revised: 07.11.14 - Accepted: 08.11.14 Published: 17.12.14
c 2014 The authors
edition of the Diagnostic and Statistical Manual of Mental Disorders (DSM-V) (American Psychiatric Association, 2013), one
of the criteria necessary for diagnosing Autism Spectrum Disorder (ASD) is an impairment in social communication and social interaction, including deficits in social-emotional reciprocity,
deficits in non-verbal communicative behaviours used for social
interaction and deficits in developing and maintaining relationships. There are two main theories which attempt to explain the
social deficits in ASD, namely Theory of Mind and social motivation theory. Previous studies have supported the hypothesis that
Theory of Mind is impaired in children with ASD (Baron-Cohen,
2000). Theory of Mind is required for the understanding of the
mental states of others, including their beliefs, desires, intentions
and emotions (Baron-Cohen, 2001). In other words, children with
ASD find it challenging to infer the thoughts of others. An impairment in Theory of Mind would result in difficulties during
social communication (Cummings, 2009). In contrast, some theories have suggested that the social communication deficit arises
from lack of social motivation rather than an impairment in social
cognition. Social motivation models indicate that due to their
impairment in early social attention, the social learning experience of children with ASD would be limited during development.
Therefore, as a result of their preference for non-social stimuli,
the impairment in social skills and social cognitive development
would be more significant (Brodkin et al., 2012).
Social skills intervention must be part of the therapeutic programme of children with ASD since it is unlikely that these skills
will improve spontaneously (Flood et al., 2010). Furthermore, the
impairment in interacting with others limits these children’s development of additional social communication abilities. Intervention targeting social communication should focus on encouraging
the acquisition of skills, enhancing performance of skills, eliminating competing negative behaviours and facilitating generalisation
(Gresham, Horner & Sugai, 2001). According to Forness et al.
(1999), intervention is more effective when it targets specific skills
rather than when it is general. Moreover, frequent therapy sessions would produce more positive results (Gresham et al., 2001).
Social communication skills are generally targeted using a behavioural approach which involves modelling, prompting and reinforcement (Baker, 2010). These strategies could be implemented
in a Social Communication Group (SCG) programme. Intervention within a small group of individuals having social communication difficulties would give them an opportunity to interact during
conversations and games while enabling them to form friendships
(Baker, 2010). Generalisation in children with ASD does not occur spontaneously and must be incorporated as part of the therapy
programme (Flood et al., 2010).
The effectiveness of social communication groups in mainstream schools
Table 1. Age and language
l Child
Age
Primary
l
(years) Language
1
8;00
English
2
8;01
Maltese
3
4
8;05
9;01
English
Maltese
5
9;02
English
6
9;03
Maltese
abilities of each child.
Summary of Language Abilities
Adequate receptive and expressive language
Receptive Language: requires modelling to understand instructions and
is unable to follow a conversation
Expressive Language: does not use complex sentences when speaking
Adequate receptive and expressive language
Receptive Language: difficulty understanding at conversational level
Expressive Language: difficulty expressing himself when answering questions
Receptive Language: difficulty understanding at conversational level
Expressive Language: adequate
Receptive Language: difficulty understanding when asked a question,
especially at conversational level
Expressive Language: difficulty expressing himself,
especially in statements requiring reasoning
Research on SCGs for children with ASD has shown inconsistent results due to the diverse presentation of participants as well
as the different methods and measurements used across studies
(e.g. Barry et al., 2003; Chung et al., 2007; Dittner et al., 2006;
Fombonne et al., 2007; Jones et al., 2004). Evidence regarding
the effectiveness of SCGs is necessary to prove that resources are
being put to good use (Cicchetti et al., 2009). Research should
aim to establish an intervention method suitable for individuals
with ASD to avoid treatments with additional risks (Offit, 2008).
The purpose of the current study was to investigate the effectiveness of SCGs for children with ASD who attend mainstream
schools. A case study approach was adopted, thus enabling a detailed group evaluation with reference to individual characteristics
of each child. Apart from identifying the specific communication
skills that were enhanced in children after participating in the
SCG programme, the study evaluates the factors which might
have affected improvement on an individual basis. It also seeks
evidence of generalisation of the skills acquired during the sessions
to everyday settings.
2
2.1
Methods
Participants
The subjects were recruited from the caseload of speechlanguage pathologists (SLPs) working in a local Health Centre.
The sample consisted of six students who had been diagnosed
with ASD and attended a mainstream school, where they had a
Learning Support Assistant (LSA). The participants were aged
between 8;00 and 9;03 years, with a mean age of 8;06 years. This
age range was selected since the chosen SCG programme, designed
by Agius (2007), was formulated for children of this age. The six
participants are referred to in the present text using a number
from 1 to 6, with Child 1 being the youngest child. Although the
participant selection criteria did not include gender specifications,
the children whose parents consented to their participation in the
study were all males.
2.2
41
Procedure
The main goals of the programme were to enhance basic skills
such as attention, listening and greeting, to improve the participants’ conversation skills, to enable them to be aware of different
emotions, to enhance their friendship skills and to encourage them
to take part in activities requiring teamwork. The programme
consisted of i) an initial meeting with the parents during which
they were provided with detailed information on the programme
http://dx.medra.org/10.14614/COMMAUT.2.40
ii) 10 group intervention sessions with the six participants and iii)
a final meeting with the parents and LSAs during which they were
given reports summarising each child’s progress. The intervention
sessions were carried out at the participants’ school, which was
considered to be a clinical setting for the purposes of this study.
Parents and LSAs were advised to attend the sessions, which took
place once a week and lasted one hour. Each session targeted a
specific set of social skills. Since the children were bilingual, both
Maltese and English were used when targeting the SCG goals.
Across sessions, the participants were provided with a series of
worksheets, which formed part of the SCG programme. Parents
and LSAs were advised to use these at home and at school to encourage generalisation of skills targeted during therapy.
A detailed overview of the programme construction is found
in Agius (2007), who describes how the activities for each session
were planned following a review of relevant literature and research
into different social skills programmes (e.g. Rinaldi, 1992; Aarons
and Gittens, 1998). Available resources such as handouts were
included among the session materials (e.g. Schroeder, 1996; Barratt et al., 2000; Mortimer et al., 2005). Emotions were addressed
during every session using The Transporters, a series of short animated films produced by the Autism Research Centre at Cambridge University (2006). Finally, the guidelines of the programme
were adapted from Rinaldi’s (1992) Social Use of Language Programme.
2.3
Measures
The effectiveness of the intervention programme was measured
using pre- and post- treatment comparison. The programme’s
Observation Schedule and Checklist (refer to Appendix 1) was
used to measure the children’s performance in various social
communication skills.
The Checklist content was based on
research into the various areas typically targeted during therapy
with children with ASD, as well as other social skills programmes
and observation schedules (see Agius, 2007). In completing
the Checklist, the children’s social communication skills were
assigned a ‘pre-rating’ and ‘post-rating’ score before and after
therapy. Table 2 exemplifies use of the Checklist rating scale with
the conversational skill of ‘Looking (Eye Contact)’ (Section 1).
Baseline and outcome measures were supplemented by field
notes on individual participants’ performance on tasks, based on
ongoing observation and evaluation. The pre- and post-treatment
measures together with the field notes were collected by the first
author. In addition, LSAs were requested to complete a feedback
form during each session, thus providing another ongoing measure
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
42
Table 2. Rating of ‘Looking (Eye Contact)’ on the Observation Schedule and Checklist.
Rating Level
1
2
3
Description
Not evident
Emerging, but need improvement
Appropriate level
Since children with ASD find it difficult to generalise skills
acquired during therapy to other settings such as home and
classroom (Keen & Rodger, 2012), the effectiveness of the
programme at home and at school was also considered. The
parents and LSAs of the participants were requested to fill in
a Questionnaire on their opinion of the child’s communication
skills before the therapy programme (refer to Appendix 2).
An identical Questionnaire was completed upon completion of
intervention. Both the Checklist and the Questionnaire were
divided into five main sections evaluating conversation skills,
emotions, games, teamwork and friendship skills.
Reliability and validity of measures were not addressed directly
in the methodological design due to time restrictions. However,
use of the Checklist in previous research (Agius, 2007) showed
that it effectively evaluated skills targeted during programme
sessions. Moreover, it was explicitly designed to accompany this
particular therapy programme. The specificity of measures to
the participants reduced the generalisability of results, limiting
their external validity (Jewell, 2014). Measurement error in preand post-treatment scores was minimised since the same observer
assigned all ratings. The Questionnaire items targeted the same
skills addressed in the Observation Schedule and Checklist,
enhancing the validity of results. However, completion of the
Questionnaire by different individuals may have introduced
observer bias in the responses.
Example
No eye contact
Eye contact inconsistent
Eye contact present even
during conversations
children was calculated by obtaining a percentage of the total
number of enhanced skills in relation to the skills that were not
at an appropriate level pre-therapy. This is represented in Figure
1.
Figure 1: Overall percentage improvement of all children
compared to the mean.
Data collected were analysed using both quantitative and qualitative methods. Data obtained from the Checklist and Questionnaire was analysed quantitatively using descriptive statistics. The
ongoing observations of children’s performance during tasks and
the feedback forms completed by the LSAs for every session were
analysed qualitatively.
2.4
Ethical considerations
Ethical approval from the University of Malta’s Research
Ethics Committee was obtained prior to data collection. The
Committee’s ethical guidelines were followed throughout the
research study.
3
3.1
Results
Analysis of Performance within the
Clinical Setting
Of the 10 sessions making up the SCG programme, each child
attended at least eight. Average attendance was 92% of the total
number of sessions. The average attendance of relatives (mothers
and grandmothers) and LSAs was 66% and 90% respectively.
Pre- and post-rating Checklist scores were compared to
calculate the percentage improvement in social communication
skills for each child and the mean percentage improvement for the
group. Skills not requiring further intervention at the pre-therapy
stage were identified through the Checklist. Skills which were
not evident or were still emerging at the onset of therapy were
grouped according to whether they improved or not by the end
of the programme. Improvement was accounted for when skill
ratings moved up by one or two ranking points. The overall
improvement of each child and the mean improvement of all six
http://dx.medra.org/10.14614/COMMAUT.2.40
Figure 2: Mean percentage improvement of the main
areas of social communication skills.
The mean percentage improvement of the six participants in
conversation skills, emotions, games, teamwork and friendships
skills was calculated and represented in Figure 2. A qualitative
description of relevant results is provided in the next sections.
3.1.1
Conversation Skills
The main conversation skills targeted during the SCG included
greetings, eye contact, turn taking, listening, topic maintenance,
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
initiation and voice volume.
- Greetings: The six children all showed improvement in greeting by saying “hello” and “goodbye” more spontaneously.
- Eye contact: None of the children showed adequate eye contact for conversational purposes pre-therapy, but 50% of them
improved this skill. Child 4, 5 and 6 improved by maintaining
more eye contact. Child 1 was able to maintain eye contact when
listening but not when speaking. Child 2 avoided looking at the
listener’s face when speaking so that eye contact was not evident
whereas Child 3 made eye contact when greeting but not during
conversation.
- Turn taking: Five of the children did not show appropriate turn taking skills pre-therapy and only Child 4 did not show
improvement since he did not wait for his turn to speak during
conversation.
- Listening: Five of the children presented with difficulties
in listening attentively during conversation since they interrupted
the speaker or appeared to be distracted. Child 4 was the only
child who did not show improvement in active listening due to his
tendency to vocalise when someone else was speaking.
- Topic maintenance: Improvement in topic maintenance was
observed in all the children since they were able to speak for a
longer time about a particular subject.
- Initiation: The six children presented with difficulties in initiating a conversation. Five of them showed improvement by introducing a topic or by asking questions.
- Volume: Of the four children who presented with an inappropriate volume level, three showed improvement. Child 2 increased
the volume level of his voice while Child 4 and 6 decreased their
loudness. Child 5 did not use adequate volume since it was either
too high or too low.
3.1.2
Emotions
The emotions targeted were happy, sad, angry, afraid, excited,
worried, surprised, disappointed, tired, unfriendly, sorry, kind
and helpful. Five of the participants had presented with difficulties in explaining and/or recognising emotions in themselves pretherapy. They all showed improvement in this area. In addition,
all six children presented with difficulties in explaining and/or
recognising emotions in others. Only Child 2 did not show any
evident improvement in the latter. By the end of the programme,
50% of the children, including Child 1, 3 and 5, also exhibited an
understanding of complex emotions.
3.1.3
Games
Games included in the SCG focused on enhancing participation, following and accepting rules, attending as well as showing
appropriate behaviour when winning or losing. Five of the children presented with difficulties in participation during games and
an improvement was observed in that they became more attentive. This was evidenced by an increase in their listening and/or
looking behaviours. All of the children showed an improvement
in collaboration during games, which was observed through turn
taking, understanding and/or following instructions and accepting and/or following rules. Although all the children behaved
appropriately when winning a game, four of them had difficulties
when losing. Three of the latter showed improvement in behaving
appropriately in this area.
3.1.4
Teamwork
Teamwork activities included in the SCG focused on encouraging collaboration, sharing, commenting and giving assistance.
Although the children were encouraged to work together, two of
them preferred working alone throughout all the sessions. Improvement was noted in the skills listed below.
- Collaboration: The children who tended to take a leadership
role improved by allowing others to contribute, while those who
preferred working alone improved by joining the rest of the group.
http://dx.medra.org/10.14614/COMMAUT.2.40
43
Child 2 showed progress by simply participating, given that in earlier sessions he refused to take part in teamwork activities. Child
6 was the only participant who did not demonstrate any difference
in collaboration pre- and post-therapy since he preferred working
individually.
- Sharing materials: Four of the children were not observed to
share materials pre-therapy. Three of them improved by sharing
the materials used during the crafts activities with the members
of the team. This was not observed in Child 2.
- Commenting: Discussion of ideas was not evident in the
initial session. Four of the children improved since they started
discussing ideas with the group while the others just followed the
rest of the team.
- Giving assistance: Only one of the children was observed
helping others during the initial session. However, by the final
session, three other children started giving assistance by helping
others find pictures, helping them to finish their puzzle or preparing for the activities.
- Leadership: Although not targeted directly, three out of the
six children improved in leadership skills by accepting leadership,
speaking for the team and accepting ideas of others.
3.1.5
Friendship skills
Friendships skills targeted through role play and brainstorming
included asking another child to play, asking a question, introducing oneself, controlling anger, practising independence and solving
problems. Improvement observed in friendship skills is described
below.
- Welcoming others: The participants became more welcoming towards their peers as the sessions progressed, except for Child
5. This trend was evident as participants accepted peers joining
them when asked to play with them. Willingness to work with
others was also shown during teamwork activities. Participants
also started showing a preference for particular peers when choosing teams.
-Introducing self : All children improved in introducing themselves by using greetings, saying their name and their age, and
mentioning their hobbies.
- Reciprocity: Four of the participants took part in more reciprocal conversations. However, this improvement was not observed in Child 1, who continued talking in a monologic style
most of the time, and in Child 2, who did not take part in conversations.
- Controlling Anger: Child 4, 5 and 6 became more able to
control their anger when waiting for their turn during games and
when disagreeing with a peer. Child 1 and 2, who also presented
with difficulties in controlling their anger, did not show any improvement in this regard.
- Independence: Child 1, 3 and 5 became more independent
when making decisions while the other participants did not show
any improvement in this area.
- Solving problems: Although all of the children presented
with difficulties in solving problems during social situations, only
Child 1 and 3 showed progress in this skill.
- Making friends: All the children improved in making friends
except for Child 2 who, even though frequently encouraged, was
rarely seen interacting with the others.
3.2
Evaluation of Performance at Home
and at School
The pre-therapy and post-therapy Questionnaires given to parents and LSAs were compared. Figures 3 and 4 show percentage
change in rating of skills according to whether parents and LSAs
reported improvement, no change or regression. The mean percentage improvement of skills at home and at school was 20%
and 19% respectively. Most of the skills remained the same after
therapy, both at home and at school. According to the Ques-
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
tionnaire data, regression was evident in some of the skills. This
outcome is discussed further in the next section. The percentage
improvement at home of Child 2 is not included since the parents’
post-therapy Questionnaire was not returned.
Figure 3: Percentage improvement at home relative to
the mean percentage improvement for the whole group.
losing. These variables might therefore account, at least in part,
for lack of improvement in the skills targeted by the programme.
However, it could not be established whether limited progress was
specific to children with ASD since a control group of typicallydeveloping children was not included in the study.
Attendance did not seem to affect the children’s individual
improvement. However, it might have had an impact if they
missed more than 80% of the sessions. More improvement was
shown in children who a) were English-speaking, b) had more
age-appropriate skills before therapy and c) had adequate understanding at conversational level. English-speaking children possibly showed most improvement since they tended to participate
and lead more in activities, resulting in more use of English during sessions which in turn encouraged their participation further.
Nevertheless, this improvement could be a result of their increased
willingness to participate, which led to more opportunities for
practising their skills. Another factor which might have had an
impact on this result is the fact that the DVDs targeting emotions were in English. Although Maltese was also used during
the sessions, the use of English in the previously mentioned task
might have been a limitation for the Maltese-speaking children.
Those children having a more positive pre-therapy status showed
increased improvement since they presented with fewer skills requiring intervention. Another possible reason is that they had
more skills which were already emerging pre-therapy, rather than
skills which were not evident. Therefore, they required intervention on improvement of skills as opposed to others who necessitated intervention on acquisition. Finally, the comprehension difficulties exhibited by Child 2, 4, 5 and 6 seemed to have affected
performance during tasks requiring conversational understanding,
namely free conversation with peers, show-and-tell and teamwork
activities. Improvement in each of the main skills addressed by
the SCG programme is discussed in the following sections.
4.1
Figure 4: Percentage improvement at school relative to
the mean percentage improvement for the whole group.
4
Discussion
The purpose of this study was to evaluate improvement in the
social communication skills of six children with ASD after attending a 10-week SCG programme in a mainstream school. Based
on observations within the clinical setting and parents’ and LSAs’
ratings, improvement was evident in the social communication of
all six children taking part in the study. However, a 100% improvement was not shown by any of the children, implying that further
therapy on social communication was required. Social interaction
and pragmatic skills are influenced by cultural factors (Bratanić,
2007; Harzing & Neyer, 2008). It should therefore be taken into
consideration that difficulties in social communication might also
be observed in typically-developing school-aged children, as in the
inability to wait for a turn. In addition, personality has an impact
on reaction to social situations even in typically-developing children (Mischel & Shoda, 2008), as in acceptance or otherwise of
http://dx.medra.org/10.14614/COMMAUT.2.40
44
Conversation skills
The results obtained for conversation skills correspond to those
reported in the study by Barry et al. (2003), where improvement
was mostly shown in greetings rather than in other conversation
skills. However, Barry et al. (2003) did not evaluate improvement
in specific conversation skills such as eye contact, turn taking, listening, topic maintenance, initiation and voice volume. In Cragar
and Horvath’s (2003) case study of a 10-year-old boy attending
group therapy, improvement in conversations was evident in that
the child interrupted the speaker less. Yet, lack of progress in the
child’s understanding of non-literal language and humour was reported, similar to findings emerging from the Checklist and Questionnaire data in the current investigation. Several studies have
reported an improvement in the initiation of social interactions as
a result of group therapy (e.g. Bauminger, 2002; Crooke, Hendrix
& Rachman, 2008; Kroeger, Newsome & Schultz, 2007). In this
study, only Child 2 did not improve in initiating conversations.
At the start of the programme, this child did not participate in
interactions, so that for him, progress entailed maintaining a conversation rather than initiating it. In the four case studies evaluated by Chung et al. (2007), all children progressed when making
initiating comments. However, in contrast to the current study,
Chung et al. (2007) also involved typically-developing peers who
modelled adequate use of social skills to the participant group.
4.1.1
Emotions
The children showed more improvement in recognising and explaining emotions as experienced by themselves rather than by
others. This ties in with the proposed difficulties in Theory of
Mind (Baron-Cohen, 2000), which prevents children with ASD
from understanding the perspective of others, including their feelings. Improvement in the understanding of complex emotions was
only observed in three of the children. This was expected since
studies such as Balconi and Carrera’s (2007) reported that chil-
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
dren with ASD had the most difficulty in recognising complex
emotions. Baron-Cohen et al. (2007) found that difficulties in
recognising complex emotions might persist through adulthood.
However, there is evidence that intervention may encourage the
development of these emotions. In fact, Bauminger’s (2002) study
on the effectiveness of cognitive behavioural intervention for children with ASD in a group setting reported improvement in the
production of complex emotions.
4.2
Games
The highest percentage improvement in skills was observed in
games. This was probably due to the fact that games were more
structured when compared to the tasks used to target the other
objectives. In addition, the prospect of winning acted as a motivator, which reinforced the children’s participation and collaboration during games. This aspect of reinforcement forms part of
the behavioural approach which encourages learning in children
with ASD (Baker, 2010). There are very few studies that have
evaluated the effectiveness of games as part of a SCG programme.
Barry et al. (2003) found that children showed more improvement
in play skills compared to conversation skills, while Dittner et al.
(2006) reported that children improved in their ability to play
with others. Targeting social skills through games, Jones et al.
(2004) observed improvement in all the skills addressed, although
progress in the children’s actual performance during games was
not evaluated.
4.3
Teamwork
There was a general improvement in collaboration, sharing materials, commenting, giving assistance and leadership. This corroborates the findings of Jones et al.’s (2004) study, in which participants were reported to start offering help and encouragement
to others. An improvement in the sharing of ideas was observed in
four of the children in the current study. Improvement in this area
was similarly reported by Jones et al. (2004). Leadership skills
were observed in the children who were more willing to collaborate in the earlier sessions of the programme. Dittner et al. (2006)
found no significant improvement in leadership skills in their investigation. However, the scales they used to assess performance
might not have been sufficiently valid since they included general social skills and did not evaluate the abilities targeted during
sessions.
4.4
Friendship skills
Friendship skills exhibited the least improvement, possibly because the relevant intervention was less structured and more unpredictable, excluding the friendship skills targeted during brainstorming activities and role play. The lack of structure in intervention was due to the fact that it depended on situations presenting
themselves during the sessions, which would differ from one group
to another. Five of the participants improved in making friends,
but Child 2 failed to interact with others through to the final
session. In Fombonne et al.’s (2007) study, 12 out of the 13 adolescents with Asperger’s Syndrome and High-Functioning Autism
who gave feedback on their ability to make friends reported an
improvement. However, this outcome was based on self-report
rather than the researchers’ observations, suggesting that the participants’ subjectivity in reporting may have influenced results. In
Barnhill et al.’s (2002) study, seven male adolescents were able to
form a friendship with one other group member, even though they
did not improve in the actual goals targeted during therapy. Cragar and Horvath (2003) reported that the child observed in their
case study managed to make friends at school after attending a social skills group. Moreover, in a parent-assisted social skills group
investigated by Dillon et al. (2012), 28 teenagers were successful in
maintaining most of the friendship skills acquired during therapy.
The current study was unable to determine whether friendships
http://dx.medra.org/10.14614/COMMAUT.2.40
45
were maintained since a follow-up assessment was not carried out
due to a limited time frame.
4.5
Generalisation
The limited evidence of generalisation could have resulted from
the lack of implementation of worksheet activities at home or at
school. Although parents and LSAs were encouraged to use these
worksheets, it was reported that they did not have time to implement them. Most children with ASD are not able to generalise
skills outside the clinical setting without additional support, suggesting that this aspect must form an integral part of any SCG
programme (Flood et al., 2010). Previous studies have also reported limited generalisation skills. For example, Barry et al.
(2003) found greetings to be generalised as opposed to conversation and play skills. Generalisation was reported by the parents
taking part in Fombonne et al.’s (2007) study, but no evidence
of generalisation was present in the investigation carried out by
Jones et al. (2004). A decrease in aggression, increased selfcontrol and improved interaction were reported by Dittner at al.
(2006). In the latter study, however, parents participated directly
during sessions and the measurement scales were based on their
reports. Cragar and Horvath (2003) reported an increase in generalisation after a nine-month follow up.
When comparing pre- and post-therapy Questionnaires completed by the parents and LSAs, regression was observed in specific skills which differed across participants. This outcome could
have been due to a change in the parents’ and LSAs’ perceptions of
the children’s skills. In turn, the latter could have resulted from a
recent incident involving the child, such as difficulties with conflict
resolution emerging during an argument with a peer. Regression
might have also been an outcome of parents’ and LSAs’ increased
awareness of social communication deficits: upon completion of
the programme, they were likely to be more knowledgeable in the
requirements of each skill. Another possibility could be that parents and LSAs compared their child with other participants in
the group and subsequently rated his skills as less appropriate.
In addition, the pre-therapy Questionnaire was completed at the
beginning of the scholastic year, when LSAs might have not yet
had sufficient experience with the children to judge whether specific skills were at an appropriate level pre-therapy. Furthermore,
reporting bias in parents and LSAs may have resulted in lack of
objectivity in the generalisation data. The latter was manifested
in discrepancies in the ratings of individual children’s behaviours
by the two observers.
4.6
Limitations
The findings of this study cannot be generalised to the broader
population of children with ASD due to the small sample size.
The data were obtained from a case study of a single group, so
that the results are not necessarily applicable to other Maltese
children attending a different mainstream school. Regretfully,
the study lacked a control group consisting of children not receiving group therapy. This would have allowed comparison with
the experimental group, providing more objective results regarding the effectiveness of the SCG. However, the ethical issue of
limiting therapy to the experimental group led to exclusion of a
control group from the study’s methodological design. Nonetheless, improvements in social communication do not usually occur spontaneously (Flood et al., 2010), which implies that unless
the participants were attending additional therapy, improvement
shown could be attributed to the SCG. Indeed, at the beginning
of the programme, none of the parents reported that their children were receiving intervention targeting their social skills. It
is also acknowledged that the Checklist and Questionnaire measures depended on the perspective of the individuals completing
these instruments. Yet, subjectivity should have been minimised
since forms were completed by the same individuals pre- and post-
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
therapy. Ensuring the validity and reliability of the Checklist and
Questionnaire data would have increased the objectivity and accuracy of results. In addition, skills emerging during the group
sessions might have been restricted by the structured nature of
the clinical setting, limiting the representativeness of the data.
5
Conclusion
The results of this study support the claims in the literature
that SCGs are effective in enhancing the social communication
skills of verbal children with ASD. The percentage improvement
resulting in the main skills targeted highlights the importance of
structured activities, motivation through rewards and repetition
of tasks for effectiveness in the intervention of children with ASD.
Although differences in children’s primary language, pre-therapy
status and understanding at conversational level had an impact
on their individual improvement, further investigation is required
regarding the effects of these specific factors. Results showed limited evidence of generalisation, but the parents and LSAs still
reported an improvement in each child’s specific deficits. These
positive results should encourage the implementation of SCGs in
mainstream schools, although further research on their effectiveness with a more representative sample is recommended.
6
Acknowledgements
We would like to thank the participants, their families and their
LSAs for taking part in the study.
7
Funding
This research has received no specific grant from any funding
agency in the public, commercial or non-profit sector.
8
Conflicts of interest
The authors report no conflicts of interest.
References
Agius, K. (2007) Social Communication Groups in a Mainstream
School with Children with Autistic Spectrum Disorders. Unpublished M.Ed. Dissertation. United Kingdom: University of Birmingham.
American Psychiatric Association (2013) Diagnostic and Statistical Manual of Mental Disorders (5th ed.). Arlington, VA:
American Psychiatric Publishing.
Baker J. (2010) Social skills training for children on the autism
spectrum: current research and integration. Autism Advocate, 58(1), pp. 8-13.
Balconi, M. & Carrera, A. (2007) Emotional representation in facial expression and script: a comparison between normal
and autistic children. Research in Developmental Disabilities, 28, pp. 409-422. doi: 10.1016/j.ridd.2006.05.001
Barnhill, G. P., Cook, K. T., Myles, B. S. & Tebbenkamp, K.
(2002) The effectiveness of social skills intervention targeting nonverbal communication for adolescents with Asperger
syndrome and related pervasive developmental delays. Focus on Autism and other Developmental Disabilities, 17(2),
pp. 112-118.
Baron-Cohen, S. (2000). Theory of Mind and autism: a fifteen year review. In D. J. Cohen, H. Tager-Flusberg & S.
Baron-Cohen (Eds) Understanding Other Minds: Perspectives from developmental cognitive neuroscience (pp. 3-20).
Oxford, U.K.: Oxford University Press.
Baron-Cohen, S. (2001) Theory of Mind in normal development
and autism. Prisme, 34, pp. 174-183.
Baron-Cohen, S., Golan, O., Hill, J. J. & Rutherford, M. D.
http://dx.medra.org/10.14614/COMMAUT.2.40
46
(2007) The ’Reading the mind in the voice’ test-revised:
a study of complex emotion recognition in adults with and
without autism spectrum conditions. Journal of Autism
and Developmental Disorders, 37, pp. 1096-1106. doi:
10.1007/s10803-006-0252-5.
Barry, T. D., Bodin, S. D., Gilmore, T., Klinger, L. G., Lee,
J. M. & Palardy, N. (2003) Examining the effectiveness
of an outpatient clinic-based social skills group for highfunctioning children with autism. Journal of Autism and
Developmental Disorders, 33(6), pp. 1096-1106. doi: 01623257/03/1200-0685/0.
Bauminger, N. (2002) The facilitation of social-emotional understanding and social interaction in high-functioning children
with autism: intervention outcomes. Journal of Autism and
Developmental Disorders, 32(4), pp. 283-298.
Bratanić, M. (2007) Nonverbal communication as a factor in linguistic and cultural miscommunication. In A. Esposito, E.
Keller, M. Bratanić & M. Marinaro (Eds) Fundamentals
of Verbal and Nonverbal Communication and the Biometric Issue (pp. 82-91). Amsterdam, The Netherlands: IOS
Press.
Brodkin, E. S., Chevallier, C., Kohls, G., Schultz, R. T. &
Troiani, V. (2012) The social motivation theory of autism.
Trends of Cognitive Sciences, 16(4), pp. 231-239. doi:
10.1016/j.tics.2012.02.007.
Chung, K., Drewry, J., Matthews, T., Mosconi, M., Reavis, S.
& Tasse’, M. J. (2007) Peer-mediated social skills training
program for young children with high-functioning autism.
Research in Developmental Disabilities, 28, pp. 423-436.
doi:10.1016/j.ridd.2006.05.002.
Cicchetti, D., De Los Reyes, A., Klin, A., Koenig, K. & Scahill, L.
(2009) Group intervention to promote social skills in schoolage children with pervasive developmental disorders: reconsidering efficacy. Journal of Autism and Developmental
Disorders, 39, pp. 1163-1172. doi: 10.1007/s10803-0090728-1.
Cragar, D. E. & Horvath, L. S. (2003) The application of social
skills training in the treatment of a child with Asperger’s
disorder. Clinical Case Studies, 2(1), pp. 34-39. doi:
10.1177/1534650102239087.
Crooke, P., Hendrix, R. & Rachman, J. (2008) Brief reports:
measuring the effectiveness of teaching social thinking to
children with asperger syndrome (AS) and high-functioning
autism (HFA). Journal of Autism and Developmental Disorders, 38, pp. 581-591.
Cummings, L. (2009) Clinical Pragmatics. Cambridge, U.K.:
Cambridge University Press.
Dillon, A. R., Frankel, F., Gantman, A., Laugeson, E. A. & Mogil,
C. (2012) Parent-assisted social skills training to improve
friendships in teens with autism spectrum disorders. Journal of Autism and Developmental Disorders, 42, pp. 10251036. doi: 10.1007/s10803-011-1339-1.
Dittner, C. A., Mellon, M., Sim, L. & Whiteside, S. P. (2006) Effectiveness of a social skills training program with school age
children: transition to the clinical setting. Journal of Child
and Family Studies, 15, pp. 409-418. doi: 10.1007/s10826006-9049-6.
Flood, M. F., Hansen, D. J., Jordan, J. A. & Smith, A. J. (2010)
Social skills interventions. In C. A. Erdley, D. J. Hansen,
D. W. Nagle & P. J. Norton (Eds) Practitioner’s Guide to
Empirically Based Measures of Social Skills (pp. 99-115).
New York: Springer.
Fombonne, E., Meng, L., Tagalakis, V., Tse, J. & Strulovitch, J.
(2007) Social skills training for adolescents with asperger
syndrome and high-functioning autism. Journal of Autism
and Developmental Disorders, 37, pp. 1960-1968. doi:
10.1007/s10803-006-0343-3.
Forness, S. R., Kavale, K. A., Mathur, S. R., Quinn, M. M. &
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
47
Rutherford, R. B. (1999) A meta-analysis of social skill interventions for students with emotional or behavioral disorders. Journal of Autism and Developmental Disorders,
7(1), pp. 54-64.
Gresham, F. M., Horner, R. H. & Sugai, G. (2001) Interpreting
outcomes of social skills training for students with highincidence disabilities. The Council for Exceptional Children, 67(3), pp. 331-334.
Harzing, A. W. & Neyer, A. K. (2008) The impact of culture on interactions: five lessons learned from the European Commission. European Management Journal, 26(5), pp. 325-334.
doi: http://dx.doi.org/10.1016/j.emj.2008.05.005
Jewell, D. V. (2014) Guide to Evidence-based Physical Therapist
Practice. Sudbury, MA: Jones & Bartlett.
Jones, W. P., Miller, S. P., Pierce, T. B., Strawser, S. & Webb,
B. J. (2004) Effects of social skill instruction for highfunctioning adolescents with autism spectrum disorders.
Focus on Autism and other Developmental Disabilities, 19,
pp. 53-62. doi: 10.1177/10883576040190010701.
Keen, D. & Rodger, S. (2012) Working with Parents of a Newly
Diagnosed Child with an Autism Spectrum Disorder: A
guide for professionals. London, U.K.: Jessica Kingsley.
Kroeger, K. A., Newsome, C. & Schultz, J. R. (2007) A comparison of two group-delivered social skills programs for young
children with autism. Journal of Autism and Developmental Disorders, 37, pp. 808-817.
Mischel, W. & Shoda, Y. (2008) Towards a unified theory of personality: integrating dispositions and processing dynamics
within the cognitive-affective processing system. In L. A.
Pervin, O. P. John & R. W. Robins (Eds) Handbook of Personality: Theory and research (pp. 208-241). New York:
The Guilford Press.
Offit, P. (2008) Autism’s False Prophets: Bad science, risky
medicine, and the search for a cure. New York: Columbia
University Press.
Prizant, B. M. & Wetherby, A. M. (2005) Critical issues in enhancing communication abilities for persons with autism spectrum disorders. In A. Klin, D. Cohen, F. R. Volkmar &
R. Paul (Eds) Handbook of Autism and Pervasive Developmental Disorders - Volume Two: Assessment, interventions, and policy (3rd ed.). Hoboken, N.J.: John Wiley &
Sons.
http://dx.medra.org/10.14614/COMMAUT.2.40
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
48
Appendix 1
http://dx.medra.org/10.14614/COMMAUT.2.40
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
http://dx.medra.org/10.14614/COMMAUT.2.40
49
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
http://dx.medra.org/10.14614/COMMAUT.2.40
50
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
51
Appendix 2
http://dx.medra.org/10.14614/COMMAUT.2.40
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
http://dx.medra.org/10.14614/COMMAUT.2.40
52
http://www.um.edu.mt/healthsciences/mjhs
The effectiveness of social communication groups in mainstream schools
http://dx.medra.org/10.14614/COMMAUT.2.40
53
http://www.um.edu.mt/healthsciences/mjhs
Malta Journal of Health Sciences
http://www.um.edu.mt/healthsciences/mjhs
Doi: http://dx.medra.org/10.14614/EXPERAMP.2.54
Research paper
THE LIVED EXPERIENCE OF PERSONS WITH AN
AMPUTATION
Chanelle Grech, Roberta Farrugia Debono
Department of Psychology, Faculty for Social Wellbeing, University of Malta, Msida, Malta
Abstract.
This paper presents an exploratory study of
Maltese persons living with an amputation. Semi-structured
interviews were conducted with four participants. Three of the
participants had a lower limb amputation, while the fourth had
an upper limb amputation. Interpretative phenomenological
analysis (IPA) was used in the analysis phase. The three core
superordinate themes that emerged from the data revolved
around the emotional reactions towards the amputation, the
numerous changes it brought to the participants’ lives, and the
support which enabled amputees to cope with their situation.
The study highlights the range of emotions experienced by the
participants on having an amputation and the changes in their
lifestyle. Support from family, friends, health professionals and
other amputees was identified as a factor that helped participants
cope and adapt to their new condition.
in hospital and medical facilities (Government of Malta, 2011).
Various studies on the experiences of amputees (e.g. Desmond
& MacLachlan, 2006; Liu et al., 2010; Senra et al., 2012) focus
on three broad aspects: the psychological impact, the processes
of adjustment linked to the change in physical image and pain,
as well as the impact of social factors such as social support and
education. Furthermore, most of the available research (Horgan
& MacLachlan, 2004; Senra et al., 2012) focuses on the lived experience of persons having an amputation of the lower limb. This
study aims to contribute to this literature by exploring the lived
experience of Maltese individuals with a lower or upper limb amputation.
2
2.1
Keywords: amputation, lived experience, prosthesis, social support, qualitative research, interpretative phenomenological analysis
1
Introduction
Limb amputation is considered to be one of the oldest surgical
procedures with a history of more than 2,500 years (Van der Meij,
1995). It is defined as a medical procedure in which there is “partial or complete removal of a limb for a variety of reasons, including
tumours, gangrene, intractable pain, crushing injury or uncontrollable infection” (Fremgen & Frucht, 2009, p.10). Amputation is
usually considered as a last resort in the case of life endangerment
(Paudel, Shrestha & Banskota, 2005), since it brings about drastic social, economical and psychological changes to both the amputees’ and their families’ lives (Sinha, van den Heuvel & Arokiasamy, 2011). Local statistics indicate that there were approximately 88 Maltese patients who had undergone one or multiple
major amputations over a one-year time frame, between 1st June
2011 and 31st May 2012 (Cassar, 2012). Fortunately, the number
of major limb amputations above or below the knee in Maltese
patients has decreased by around 33% between 2003 and 2010, as
a result of early detection, timely intervention and improvement
Correspondence to: Chanelle Grech
(chanellegrech@gmail.com)
Received: 22.07.14 - Revised: 22.11.14 - Accepted: 28.11.14 Published: 17.12.14
c 2014 The authors
Methods
Participants
Four Maltese participants were interviewed regarding their
lived experience of being an amputee. Interpretative phenomenological research is usually conducted using small samples (three
to six participants) since a detailed analysis of each case is
necessary (Smith, Flowers & Larkin, 2009). The purposive
sample consisted of two male and two female amputees whose
ages ranged between 30 and 80 years. Participants were recruited
from the Amputees4Amputees Association, which is a support
group for Maltese amputees. They were informed about the
study by the Association through an information letter which
contained details on the nature, aims and requirements of the
study. Those persons who were willing to participate informed
the designated Association member, who then forwarded their
contact details to the first author. Criteria for inclusion in the
study included being over 18 years of age and having had an
amputation for at least 4 years prior to the study, to ensure that
emotional distress levels would have returned within the norm
(Horgan & MacLachlan, 2004). A profile of the participants is
presented in Table 1.
2.2
Data Collection
The interview schedule consisted of ten semi-structured and
open-ended questions. Questions were open-ended to provide participants with an opportunity to elaborate on their responses.
Their semi-structured nature allowed for probing and a more
conversation-like interview, rather than a rigid question-andanswer approach (Langdrige & Hagger-Johnson, 2009). The participants could opt to be interviewed in Maltese or English. In
fact, all opted for the Maltese language. The duration of the
tape-recorded interviews was 40-60 minutes and took place in a
55
The lived experience of persons with an amputation
Table 1. Participant characteristics.
Pseudonym
Gender
Status
Type of
amputation
Jessica
Female
Single
Below the knee
Maria
Joseph
Mario
Female
Male
Male
Married
Married
Married
Above the knee
Upper Limb
Below the knee
Reason
Birth defect limb malformation
Gangrene
Accident
Accident
Note: Participants’ ages are not included in order to preserve their anonymity.
Table 2. Super-ordinate themes.
Super-ordinate themes
Sub-themes
An emotional reaction
Impact on self
Feelings of significant others
A different life
Changes in lifestyle
Prosthesis and discomfort
Searching for meaning
Social connections and support
Family and friends
Professionals as a source of information and care
setting suggested by the interviewee. Three core areas were explored, namely participant pre-amputation and post-amputation
experiences, as well as external factors such as social support and
social integration. Interviews were conducted by the first author
between December 2012 and January 2013.
3.1
An Emotional Reaction
Participants gave details of their own personal reactions to the
amputation, as well as the reactions of family members and significant others.
3.1.1
2.3
Data Analysis
Interpretative phenomenological analysis (IPA) was used, following procedures outlined by Smith et al. (2009). This approach
was found to be the most suitable to examine personal experiences
as it involves a detailed analysis of each case (Smith et al., 2009),
and therefore the researcher must take on the role of an interpreter
(Smith & Osborn, 2003). IPA aims to engage with people’s reflections on their subjective experiences, paying particular attention
to what the experience was like for every participant and how s/he
made sense of it (Smith et al., 2009). Studies using IPA are often
concerned with existential issues in relation to major life-changing
or threatening events, as in the case of the loss of a limb (Smith,
2004).
2.4
Ethics
Ethical approval to conduct this study was obtained from the
University of Malta Research Ethics Committee (UREC). All participants were asked to sign a consent form prior to the study.
They were informed that they could terminate the interview at
any point, or decline to answer any question without the need to
provide a reason. Pseudonyms were assigned to each participant,
and transcripts and audio- recordings were stored in a locked cabinet to safeguard participant confidentiality.
3
Results and Discussion
Three super-ordinate themes emerged from participant accounts. These were: a) the Initial Emotional Reaction, b) a Different Life and c) Social Connections and Support. In the following
sections, each super-ordinate theme is divided into sub-themes,
which are discussed with the use of excerpts from participant accounts and compared to existing literature.
http://dx.medra.org/10.14614/EXPERAMP.2.54
Impact on self
Most of the research available on the experience of amputation
is related to one’s reaction to his or her own amputation, which
is dependent on a number of factors such as age, level (below
or above the knee/elbow) and time since amputation (Horgan &
MacLachlan, 2004).
The participants in this study described
their initial reaction to their own amputation, occurring as a consequence of an accident or gangrene, in a negative manner. Mario
explained:
“Initially it was a huge shock. I was crushed!” 1
Maria stated:
“I really cried!” 2
Similarly, participants in a study conducted by Senra et al.
(2012) expressed feelings of sadness, shock and surprise. However, Jessica, who had her limb amputated due to a malformation
in her leg (birth defect), perceived it in a positive light, as exemplified in the following excerpt.
“The iron rods around my [malformed] leg were so ugly
and so uncomfortable. In fact, the first thing I asked
her [my mother] as a child, was whether the amputation would allow me to wear heels and any kinds of
shoes. In that aspect I can say it was more of an improvement for me.” 3
This demonstrates that the cause of an amputation may influence one’s reaction towards it (Horgan & MacLachlan, 2004). Participants in this study denied experiencing any long-lasting emo1 “G~all-ewwel
kien xokk kbir. Kont imfarrak! ”
kemm bkejt”
3 “Il-~adid li kelli ma’ saqajja [l-isfurmata] kien vera ikrah u
skomdu. Infatti, l- ewwel ~aġa li staqsejtha [lil ommi], ta’ tifla
li kont, kienet jekk inkunx nista’ nilbes takkuna u żraben li rrid.
Jigifieri minn dak il-lat jiena kelli improvement.”
2 “Tg~idx
http://www.um.edu.mt/healthsciences/mjhs
56
The lived experience of persons with an amputation
tional distress such as depression and anxiety, even though psychopathological symptoms following amputation are commonly
cited in the literature (see Hamill, Carson & Dorahy, 2010). This
could be due to “heroic cheerfulness” and denial of any problems
(Thomson & Haran, 1984, cited in Horgan & MacLachlan, 2004).
Another possible reason could be the presence of sufficient support, a possible mediator between amputation and the psychological outcome (Hamill et al., 2010).
All the amputees in this study voiced their preoccupation that
the loss of a limb would affect their physical appearance. A research study by Miller and Deathe (2004) highlighted the effect
of an amputation on aesthetics. The ability to sustain femininity
and wear heels and skirts, was also of particular importance to the
females interviewed in this study. In contrast, male participants
seemed to be more concerned over the loss of their breadwinner
status.
“Even aesthetically, as a woman, I want to make sure
that it [the prosthesis] looks good, especially if I’m
wearing a dress.” 4 (Jessica)
Joseph also described how the amputated limb affected his selfpride.
“. . . it made me feel inferior, because I could no longer
be my family’s breadwinner.” 5
This concern finds support in the literature. Men place more
importance on the functional aspect of the limb and their ability to
financially provide for the family (Murray & Fox, 2002). During
each of their interviews, participants expressed similar opinions
that certain character traits such as courage, confidence and being positive helped them to cope, as exemplified by the following
excerpt.
“. . . but every human mind is different, not everyone
thinks alike. Some people get disheartened . . . There
were some who gave up and passed away within a
fortnight.” 6 (Maria)
In a study about positive adjustment to lower limb amputation,
Unwin, Kacperek & Clarke (2009) suggested that positive mood
was a strong contributor towards a good early adjustment.
3.1.2
Feelings of significant others
Participants also described the emotional responses of significant others in their lives. Maria reported how her granddaughter,
with whom she had a close relationship, reacted to the amputation. She explained how initially her granddaughter appeared
to be afraid of her, but soon realised that her grandmother was
still the same person. Jessica, who had undergone her amputation during childhood, mentioned how preoccupied her mother
was about the comments and judgements of others, especially children’s. Mario described his experience in the following way:
“. . . they [my parents] took it really badly. Especially
my father, who had never wanted me to ride a motorcycle [for fear of an accident].” 7
The emotional difficulties that parents endure as a result of
their child’s amputation were also highlighted in a case study on
child amputation (Judd, 2001). In this study, the participant
describes the distress experienced by his mother in the following
way: “Mum. . . every time I cry she cries with me.” (p.50)
4 “Anke estetikament b~al speċi mara, nara li nkun irridha
[il-prosthesis] pulita, li jekk jien ~a nilbes libsa, tidher pulita.”
5 “ . . . bdejt in~ossni inferjuri jien, g~ax speċi I’m not the
breadwinner.”
6 “. . . imma l-mo~~ tal-bniedem, mhux kul~add ja~seb l-istess.
Min jaqta’ qalbu. . . kien hemm min qata’ qalbu u fi ~mistax miet.”
7 “. . . ~aduha [il-ġenituri] bi kbira ~afna.
Speċjalment
missieri, li ma riedni qatt bil-mutur [g~ax jibża’ li jkolli xi
inċident].”
http://dx.medra.org/10.14614/EXPERAMP.2.54
3.2
A Different Life
Findings from the present study showed that living with an
amputation may bring about several changes in one’s life. The
following section provides details on the participants’ perceived
changes in their life, as a result of their amputation.
3.2.1
Changes in lifestyle
All participants had similar views on the fact that the amputation had an effect on their independence, posing certain limitations
and at times making them dependent on others.
“. . . It’s very difficult!
You have to depend on
many people. I hate it when I can’t do something
myself.” 8 (Joseph)
Changes in lifestyle were specifically cited by participants who
previously enjoyed sports and physical activities.
“. . . certain things like running, sports and football. . . The worst thing of them all is not being able
to run and play with my children.” 9 (Mario)
According to Asano et al. (2008), mobility was the second best
predictor of quality of life among individuals who had a lower limb
amputation. In this study, participants highlighted the changes
in their everyday life brought about by the amputation. Similar
to what was reported by Liu et al. (2010), three of the participants described feelings of frustration as a result of their loss of
independance and increased dependence on caregivers.
Another participant, Maria, described the change in her intimate relationship with her husband following her amputation. She
explained that her husband had a fear of illness and was greatly
upset by her amputation. This in turn affected their sexual relationship.
“It took a very long time until we were able to be sexually intimate again, as a married couple. When he
started to initiate intimacy again, I used to just cover
it [the stump].” 10
The research literature on sexuality in amputees is limited.
However, the present study corroborates the findings of a prominent investigation on the topic by Williamson and Walters (1996),
which suggested that sexual activity in a couple was influenced by
the perceptions of the spouse regarding the stump or prosthesis.
Two of the participants explained that while reactions from people whom they had dated were mostly positive, the parents of such
individuals seemed to think otherwise.
“Some mothers didn’t want their daughters to date me.
One of them even paid for her daughter to move to
Australia.” 11
Mario, however, also described how he managed to take advantage of having an amputation.
“. . . they [females] felt more comfortable approaching
me themselves, because they felt sorry for me. Rather
than having to approach them myself, they would come
to me . . . I would then choose the best one!” 12
8 “Diffiċli ~afna! Tiddependi minn ~afna nies. Jiena jdejquni
affarijiet li ma nistax nag~milhom jien.”
9 “. . . ċertu affarijiet b~al ġiri, sport u futbol. . . L-aktar li t~oss
illi ma tistax tiġri u tilg~ab mat-tfal.”
10 “Dam voldieri ma kellniex x’naqsmu flimkien b~ala
miż żewġin. Imbag~ad meta beda jerġa’ jersaq lejja, din [is- sieq
maqtug~a] kont ng~attiha.”
11 “Ftit ommijiet ma ridux lil binthom to~roġ mieg~i. Wa~da
minnhom ~allsitilha [lit-tifla] biex titlaq l-Awstralja.”
12 “. . . bdew i~ossuhom komdi iktar it-tfajliet jiġu fuqi huma,
u kienu jaqbdu mieg~i huma milli kont immur jiena fuq tfajla. . . u
nag~żel lill-a~jar wa~da! ”
http://www.um.edu.mt/healthsciences/mjhs
57
The lived experience of persons with an amputation
3.2.2
All participants described how the use of a prosthesis involved
a process of acceptance and adjustment, as demonstrated in the
following two excerpts.
“. . . you start adjusting to the prosthesis by wearing it
for half an hour. Then you start going for very short
walks.” 13 (Mario)
“. . . You must trust your body, you must trust the leg
so that you can stand on it and walk. You can’t remain
dependent on crutches or a wheelchair.” 14 (Jessica)
These comments highlight the importance of the adjustment
process of the amputee to his/her prosthetic limb. Saradjian,
Thomson and Datta (2008) showed that adjustment to a prosthesis is a personal and individual process. All the participants
had different ways of adjusting, with some even customising their
prosthesis to suit their own personal needs or wearing it initially
for short periods of time. Participants also complained about the
discomfort and increased sweating induced by wearing the prosthesis in the hot Maltese summer, as well as the burden of its
weight. The former factor was also cited in Saradjian et al.’s
(2008) study, in which amputees spoke about the sweating and
skin sores caused by wearing a prosthesis in warm temperatures.
Although the amputees in the current study did not mention any
phantom limb pain, they complained of residual pain and pain in
the lower back. Ehde et al. (2000) state that this could be due to
altered gait patterns to accomodate the prosthesis.
3.2.3
they are obsessed and blind.” 16
Prosthesis and discomfort
Searching for meaning
Participants were consistent in reporting that the amputation
had resulted in a change in how they viewed their own lives. They
explained how they had begun to appreciate life and had learned
to live on a day-to-day basis, rather than focusing on the future.
This was even more evident in those participants who had lost
their limb in an accident, as exemplified by the following statement.
“Nowadays, I don’t take anything for granted
. . . because I know I could leave here in five minutes,
a minute or a second, and my life could end, and so
your priorities do change.” 15 (Joseph)
A common question asked by all the participants was “Why
me?”, representing an effort to understand why the amputation
had happened to them. Similarly, Dossey and Dossey (1998) had
identified the need for participants in their study to arrive at an
understanding to this question. This was reported to often result
in spiritual discomfort, which could in turn lead to the complete
negation of God in some amputees (Dossey & Dossey, 1998). This
was clearly evident in Mario’s statement.
“I don’t believe in anything anymore. When I look at
people going to church nowadays, they remind me of
when we used to watch Red Indian movies, in which
people adored wood or stones. . . People go there because
13 “. . . sakemm tibda tidraha [il-prosthesis] just kemm tilbisha
toqg~od nofs sieg~a biha. Imbag~ad mixja minn hawn s’hemm u
lura . . . ”
14 “Inti trid tafda lil ġismek, trid tafda lis-sieq, li inti trid
tirfes fuqha u timxi. Ma tistax tibqa’ tiddependi fuq il-crutches,
fuq il-wheelchair.”
15 “Illum il-ġurnata ma sirt nie~u xejn for granted. . . G~ax
naf li jiena nista’ no~roġ minn hawn ~ames minuti, minuta jew
sekonda o~ra u ~ajti tispiċċa, u allura qisek il-prijoritajiet tieg~ek
se jinbidlulek.”
http://dx.medra.org/10.14614/EXPERAMP.2.54
Conversely, Maria described a very different experience.
“It [the experience of an amputation] brought me closer
to God. It made me realise God was by my side all
along.” 17
Maria goes on to explain how her experience encouraged her to
join her local parish group. A relationship with God/higher power
has been demonstrated to offer security and strength (Baldacchino
et al., 2013) and to serve as a means of positive coping. Joseph
also described how his initial reaction was that of anger towards
God. However, over time he realised that God had nothing to do
with the incident that befell him.
3.3
Social Connections and Support
Participants all highlighted the importance of social support,
be it from family and friends, from professionals and staff at hospital, or from other amputees. Furthermore, the importance of
support from family and friends was highlighted as one of the
most important aspects in their experience.
3.3.1
Family and friends
Participants did not report any change in the way they interacted with others. However, they highlighted the importance
of re-integration and remaining socially active by joining organisations, or simply by spending time with friends and family.
Williams et al. (2004) reported that this is an important aspect
of adjusting to life after the loss of a limb. As explained by Mario:
“The way you re-integrate in the first few months. . . if
you take long to re-integrate, yes, I think you’ll suffer.
I was meeting my friends and going to the disco when
I was still in a wheelchair. Maintaining your social life
is very important!” 18
Furthermore, family members also served to encourage the amputee during the rehabilitation process. For example:
“You need to let go. You need to let go of your walking aids. In fact, the more time passed, the more
I felt pressured by those around me [to trust the
prosthesis].” 19 (Jessica)
Other family members improvised ways of enabling the amputee
to cope in his/her situation, as described by Joseph:
“I was really lucky that my son was able to modify my
prosthesis. . . he attached an electric motor which helps
me a lot.” 20
Although support from family and friends is considered to be
of importance following an amputation (Liu et al., 2010), at certain times, participants felt that they were being over-protected
by their families. For instance, Jessica recounted the following
episode.
16 “Anqas g~adni nemmen f ’xejn. Meta n~ares lejn in-nies
imorru l-knisja llum il-ġurnata jien nist~ajjilhom meta konna
naraw il-films tal-Indjani, jaduraw xi injama jew ġebla. . . In-nies
imorru hemmhekk g~ax ossessjonati u g~omja.”
17 “ Ġabitni [l-esperjenza tal-amputazzjoni] aktar viċin talMulej. Ġeg~litni nirrealizza li Alla kien mieg~i l-~in kollu.”
18 “Kif tintegra l-ewwel ftit xhur. . . jekk inti ddum biex terġa’
tintegra, iva, na~seb li tbati. Jiena kont niltaqa’ ma’ s~abi u
mmur id-diskow, kont g~adni bil-wheelchair. Importanti ~afna li
ż żomm social life! ”
19 “Inti trid you let go. Li inti dawn l-aids you let go of them.
Aktar ma beda jg~addi ż-żmien iktar ~assejtha l-pressure minn
dawk ta’ madwari [biex nafda l-prosthesis].”
20 “Jiena kont lucky ~afna wkoll illi kelli t-tifel kien g~enni
biex irranġajna l-prosthesis. . . g~amilhieli bl-electric motor u
tg~inni ~afna jiġifieri.”
http://www.um.edu.mt/healthsciences/mjhs
58
The lived experience of persons with an amputation
“Last time my mum saw me climbing up the ladder, to
change a light bulb. She started panicking and asked
me to get down [due to fear of a fall].” 21
“They all give their 100% to see you satisfied. . . they
provide the support that you need. They see you as a
person and not a number.” 26 (Maria)
The participants in Hamill et al.’s (2010) investigation similarly identified this over-protective behaviour as frustrating for
amputees.
The amputees in the current study spoke about the need to
form relationships with others in the same situation as themselves.
They also highlighted the importance of being able to share and
encourage each other throughout the experience. One of the participants had been introduced to another amputee her age before
the amputation.
In their review on rehabilitation following major traumatic amputations, Eldar and Jelić (2003) mentioned the importance of
early involvement of rehabilitation health professionals who can
provide useful advice on rehabilitaion, prosthetic options and ways
on maintaining muscle strength, as well as self-care skills.
“When they introduce you to an amputee who is already
walking, you get to see the final results.” 22 (Jessica)
Similarly, Joseph explained:
“That [meeting another amputee] helped me so much,
because automatically, when you see someone who has
been in the same situation, you can’t help listening to
them.” 23
Other participants managed to meet up with other amputees
through information meetings and sessions held by local associations for amputees or when visiting the Orthotic and Prosthetics
Unit or Physiotherapy Outpatients Department following their
amputation. These experiences tie in with Liu et al.’s (2010) finding that peer support is helpful in providing relief from emotional
distress, and reducing isolation.
3.3.2
Professionals as a source of information
and care
Health professionals, such as doctors, nurses and physiotherapists, were also perceived as an important source of support.
Participants seemed to mention different aspects of support when
describing their experience in hospital. Joseph described the support that he received from hospital staff as follows:
“I cannot complain about anything. When I was in
hospital, I had good support because I had doctors,
nurses. . . they were all supportive. They were always
there to help change my dressings [following the amputation] and provided for my needs.” 24
In contrast, Maria held a very different viewpoint.
“Nothing! While in hospital I found no emotional support at all. They give you no information on the procedure, the way it will affect your life and how to cope.” 25
Despite the mixed reactions regarding the support given during
their stay in hospital, all participants agreed on the quality of
support given at the Physiotherapy Outpatients Department and
the Orthotic and Prosthetics Unit, which they had been making
use of following their amputation.
4
Limitations
The present study addresses the lacuna in local literature on the
lived experiences of persons with lower and upper limb amputations. There is, however, a need for further research which is
longitudinal in nature and explores the lived experiences of study
participants over a period time. The limited time frame for this
study posed limitations on the number of participants with whom
interviews were conducted, and on the possibility of second interviews. Also, the fact that only one of the four participants had
an upper limb amputation limits the ability of this study to delve
into the experience of amputees of the upper limb. A more diverse
sample that included more upper limb amputees would have allowed further understanding of the experiences of persons having
different types of amputation.
5
Conclusions
This study explored the lived experience of four Maltese amputees.
It identified the various reactions and changes which occur following the loss of a limb. Being specific to the Maltese population,
it contributes to a better understanding of the experiences of persons with an amputation in the local context. It also explores the
needs of amputees in Malta as well as the type of services provided.
Furthermore, it highlights the importance of family members’ and
friends’ psychological and physical support as an essential part of
individuals’ adjustment to their new life as amputees and the use
of their prostheses.
6
Acknowledgments
The authors would like to thank the amputees who gave their
time to participate in this study and who shared their experience
with them. The authors would also like to express their sincere
gratitude towards Dr Josianne Scerri for offering her invaluable
advice on an earlier version of this paper.
7
Funding
This research study has received no specific grant from any
funding agency in the public, commercial or non-profit sectors.
8
Conflicts of Interest
The authors report no conflicts of interest.
21 “L-a~~ar
darba ommi ratni tiela’ fuq sellum, biex inbiddel
bozza. Bdiet tippanikja u tg~idli biex ninżel [ma mmurx naqa’].”
22 “Inti meta jlaqqg~uk ma’ xi ~add li diġà qed jimxi, inti
qieg~da tara l-final results.”
23 “Dik g~enitni ~afna, g~ax inti qisu awtomatikament meta
tara lil xi ~add fl-istess sitwazzjoni, tismag~hom bla ma trid.”
24 “Ma nistax ingerger. Mindu kont l-isptar kelli sapport tajjeb g~ax kelli t-tobba, kelli n-nersis. . . kollha kienu ta’ sapport.
Dejjem kienu hemm biex ibiddluli l-faxex [wara l-amputazzjoni] u
jipprovduli li kelli bżonn.”
25 “Xejn! L-isptar ma sibt l-ebda sapport emozzjonali. Ma
jag~tuk l-ebda informazzjoni dwar l-operazzjoni, kif se taffettwalek
~ajtek u kif tista’ tikkowpja.”
http://dx.medra.org/10.14614/EXPERAMP.2.54
References
Asano, M., Rushton, P., Miller, W. C. & Deathe, B. A. (2008)
Predictors of quality of life among individuals who have a
lower limb amputation. Prosthetics and Orthotics International, 32(2), pp. 231-243.
Baldacchino, D., Torskenaes, K., Kalfoss, M., Borg, J., Tonna,
A., Debattista, C., Decelis, N. & Mifsud, R. (2013) Spiri26 “Kollha jag~tuk il-100% biex jarawk sodisfatta. . . jag~tuk
kull sapport li hemm bżonn. Jarawk b~ala persuna u mhux b~ala
numru.”
http://www.um.edu.mt/healthsciences/mjhs
The lived experience of persons with an amputation
59
tual coping in rehabilitation - a comparative study: part 2.
British Journal of Nursing, 22(7), pp. 402-408.
Cassar, M. (2012) National Amputee Database (June 2011 - end
of May 2012). Pieta’, Malta: Amputee Rehabilitation Unit.
Desmond, D. M. & MacLachlan, M. (2006) Affective distress and
amputation-related pain among older men with long-term,
traumatic limb amputations. Journal of Pain and Symptom
Management, 31(4), pp. 362-368.
Dossey, B. M. & Dossey, L. (1998). Body-mind-spirit: attending
to holistic care. The American Journal of Nursing, 98(8),
pp. 35-38.
Ehde M, D., Czerniecki M, J., Smith G, D., Campbell M, K.,
Edwards T, W., Jensen P, M. & Robinson R, L. (2000)
Chronic phantom sensations, phantom pain, residual limb
pain, and other regional pain after lower limb amputation.
Archives of Physical Medicine, 81, pp. 1039-1044.
Eldar, R. & Jelić, M. (2003) The association of rehabilitation and
war. Disability and Rehabilitation, 25(18), pp. 1019-1023.
Fremgen, B. F. & Frucht, S. S. (2009) Medical Terminology: A
living language (4th ed.) NJ: Prentice Hall.
Government of Malta (2011) Significant Reduction in Major Lower Limb Amputations in Malta. [Online] Available from: https://www.gov.mt/en/Government/Press%
20Releases/Pages/2011/November/30/pr2328.aspx.
[Accessed 22nd October 2014].
Hamill, R., Carson, S. & Dorahy, M. (2010) Experiences of psychosocial adjustment within 18 months of amputation: an
interpretative phenomenological analysis. Disability and
Rehabilitation, 32(9), pp. 729-740.
Horgan, O. & MacLachlan, M. (2004) Psychosocial adjustment to
lower limb amputation: a review. Disability and Rehabilitation, 26(14/15), pp. 837-850.
Judd, D. (2001) ‘To walk the last bit on my own’ - narcissistic independence or identification with good objects: issues of loss for a 13-year-old who had an amputation.
Journal of Child Psychotherapy, 27(1), pp. 47-67. doi:
10.1080/00754170010023947.
Langdrige, D. & Hagger-Johnson, G. (2009) Introduction to Research Methods and Data Analysis in Psychology. London:
Pearson Education Ltd.
Liu, F., Williams, R. M., Liu, H. E. & Chien, N.-H. (2010) The
lived experience of persons with lower extremity amputation. Journal of Clinical Nursing, 19, pp. 2152-2161.
Miller, W. C. & Deathe, A. B. (2004) A prospective study examining balance confidence among individuals with lower limb
amputation. Disability and Rehabilitation, 26(14-15), pp.
875-881.
Murray, C. D. & Fox, J. (2002) Body image and prosthesis satisfaction in the lower limb amputee. Disability and Rehabilitation, 24(17), pp. 925-931.
Paudel, B., Shrestha, B. K. & Banskota, A. K. (2005) Two faces
of major lower limb amputations. Kathmandu University
Medical Journal, 3(11), pp. 212-216.
Saradjian, A., Thomson R, A. & Datta, D. (2008) The experience
of men using an upper limb prosthesis following amputation:
positive coping and minimizing feeling different. Disability
and Rehabilitation, 30(11), pp. 871-883.
Senra, H., Oliveira, R. A., Leal, I. & Vieira, C. (2012) Beyond the
body image: a qualitative study on how adults experience
lower limb amputation. Clinical Rehabilitation, 26(2), pp.
180-191.
Sinha R, van den Heuvel, W.J. & Arokiasamy, P. (2011) Factors
affecting quality of life in lower limb amputees. Prosthetics
and Orthotics International, 35(1), pp. 90-96.
Smith, J. A. (2004) Reflecting on the development of interpretative phenomenological analysis and its contribution to qualitative research in psychology. Qualitative Research in Psychology, 1(1), pp. 39-54.
Smith, J. A., Flowers, P. & Larkin, M. (2009) Interpretative
Phenomenological Analysis: Theory, method and research.
London: Sage Publications.
Smith, J. A. & Osborn, M. (2003) Interpretative phenomenological analysis. In J.A. Smith (Ed.) Qualitative Psychology: A
practical guide to research methods. Thousand Oaks, CA:
Sage.
Unwin, J., Kacperek, L. & Clarke, C. (2009) A prospective study
of positive adjustment to lower limb amputation. Clinical
Rehabilitation, 23(11), pp. 1044-1050.
Van der Meij, W. K. N. (1995) No Leg To Stand On: Historical
relation between amputations surgery and prostheseology.
Unpublished doctoral dissertation. The Netherlands: University of Groningen.
Williams, M. R., Ehde, M. D., Smith, G. D., Czerniecki, M. J.,
Hoffman, J. A. & Robinson, R. L. (2004) A two-year longitudinal study of social support following amputation. Disability and Rehabilitation, 26(14/15), pp. 862-874.
Williamson, G. M. & Walters, A. S. (1996) Percieved impact of
limb amputation on sexual activity: a study of adult amputees. The Journal of Sex Research , 33(3), pp. 221- 230.
http://dx.medra.org/10.14614/EXPERAMP.2.54
http://www.um.edu.mt/healthsciences/mjhs
Malta Journal of Health Sciences Online
http://www.um.edu.mt/healthsciences/mjhs
Doi: http://dx.medra.org/10.14614/COCHIMP.2.60
Research paper
SPEECH AND LANGUAGE SKILLS OF MALTESE CHILDREN
WITH BILATERAL COCHLEAR IMPLANTS: THREE CASE STUDIES
Martina Galdes, Daniela Gatt
Department of Communication Therapy, Faculty of Health Sciences, University of Malta, Msida, Malta
Abstract. The purpose of this study was to document the
speech and language skills of three Maltese children with bilateral
cochlear implants. One child was simultaneously implanted and
had a hearing age (HA) of 2;10 years at the time of testing,
another was sequentially implanted and had a HA of 3;06 years,
while the third child was sequentially implanted and had a
HA of 5;03 years. Maltese standardised speech and language
assessments were used to gather information on the children’s
speech and language skills, with data being collected during
their speech and language therapy sessions. Following data
transcription and analysis, the participants’ speech and language
abilities were compared to those of their HA- and chronological
age (CA)-matched peers using available norms for Maltese
children. Additional information regarding the children’s speech
and language history prior to and post-implantation was also
collected, providing a holistic overview of the participants’ speech
and language development. Results indicated that the children
presented with speech and language delay when compared to
their CA-matched peers.
Variations across the participants
were found in specific language skills. Similarities in language
patterns were also noted, including expressive abilities in advance
of receptive skills. These findings extend the limited data on
the speech and language skills of Maltese children with cochlear
implants, with comparison to norms for typically-developing
children being a novel approach to research in this area.
Keywords speech, language, hearing loss,
dren, cochlear implants, bilingualism
1
Maltese chil-
Introduction
In order to receive language input, children mainly rely on their
hearing abilities. If the latter are impaired, a temporary or permanent hearing difficulty may result. Sensorineural hearing loss
(SNHL) is a permanent type of hearing impairment that has often led to fitting of hearing aids. However, depending on the
severity of the hearing loss, the latter may not be ideal because
Correspondence to: Martina Galdes
(martinagaldes@gmail.com)
Received: 02.09.2014 - Revised: 23.11.2014 - Accepted: 24.11.2014
- Published: 17.12.2014
c 2014 The authors
they amplify but do not clarify incoming sound for the hearingimpaired individual (Welling, 2010). Thus, in cases where the
individual is unable to comprehend spoken language through conventional hearing aids, cochlear implants (CIs) might be an alternative (Roland, 2000). Indeed, CIs are a major technological
attainment to treat deafness (Niparko et al., 2010). They allow
the development of auditory skills in naturalistic, albeit supported
environments and are able to support the acquisition of speech,
language and communication (Archbold, 2002).
Children with a unilateral CI are reported to have difficulty understanding speech in noise and localising sound (Scherf et al.,
2009). Bilateral implantation can overcome these limitations.
Various researchers (e.g. Battista & Highhouse, 2011; Dunn et
al., 2008; Litovsky et al., 2006; Sammeth, 2007; Therres, 2012)
assigned three effects to binaural listening as a result of bilateral
implantation, namely the head shadow effect, the binaural summation effect and the binaural squelch effect. In the head shadow
effect, both ears are active. The ear with the more favourable
signal-to-noise ratio (SNR) is immediately accessible and the person can selectively attend to this ear (Therres, 2012). Binaural
summation enables the individual to perceive the sounds presented
to both ears louder. Specifically, an improvement of 3 dB is known
to occur for binaural listeners, leading to better speech perception
(Therres, 2012). Furthermore, when input is received from both
ears, the brain and the auditory system will be able to combine
inputted information from both ears and produce a better central representation, providing a clear separation of the speech and
noise signals (Therres, 2012). This is referred to as the binaural
squelch effect. With these reported benefits, the cochlear implant
enhances the individual’s hearing experience which, as a result,
would help the acquisition of speech and language.
Individuals with bilateral implants are implanted either sequentially or simultaneously. Sharma et al. (2007) compared the P1
latency, an evoked potential which analyses the auditory cortical
response to auditory stimuli, in 20 children who received a bilateral implantation during the first 3;06 years of life. Ten children
had a simultaneous implantation while the rest received sequential bilateral implants. In both groups, the average P1 latency was
measured on implant activation and at two-month intervals postimplantation up to 15 months. The P1 latency for both groups
was within normal limits by 3.5 months post-implantation. This
suggests that any bilateral implantation that occurs within the
first 3;06 years takes place within a highly plastic central auditory
nervous system.
Despite the sensitive time proposed by Sharma et al. (2007),
several studies have shown that there should be no age limit for
the second implantation. Scherf et al. (2009) observed the auditory abilities and speech performance of 35 children with sequen-
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
tial bilateral CIs following three years of bilateral implant use. CIs
offered improved auditory skills to all children, even those sequentially implanted after the age of 6;00 years, although it emerged
that the latter took longer than those implanted before the age
of 6;00 years to demonstrate bilateral benefit. The older children,
that is, those implanted after the age of 6;00 years, obtained better speech recognition levels. Additionally, at two to three years
post-bilateral implantation, almost all children benefited in noisy
situations.
In another study, Kim, Kim and Jeong (2013) investigated the
functional benefits of sequential bilateral cochlear implantation in
42 children having a mean inter-stage interval of 5;06 years between the two implants. Average ages were 4;02 years at the first
implant and 9;07 years at the second implant. The participants
were grouped according to the inter-stage intervals. The second
and third group, those with an inter-stage interval of 5;00 and
6;09 years, and 7;00 and 9;09 years respectively, obtained comparable results in both quiet and noise tests to the first group, who
had a shorter interval of 3;00 and 4;09 years, two years following
bilateral use. Additionally, speech performance in noise improved
even in the older children following prolonged use of bilateral CIs.
These results show that the binaural hearing experience plays an
important role in the speech outcome.
On the other hand, simultaneous implantation enhances children’s speech and language acquisition and decreases their ability
difference in comparison to their chronological age (CA)-matched
peers. Wie (2010) investigated a group of 21 children with cochlear
implants two years post-simultaneous implantation. The majority of these acquired receptive and expressive language skills at
a faster pace than expected in comparison to their hearing age
(HA)-matched peers.
Apart from the type of implant, language skills depend on the
severity of the SNHL, the age of identification and implantation, as
well as the child’s cognitive abilities (Welling, 2010). For example,
Niparko et al. (2010), who studied 188 children who underwent
cochlear implantation before the age of five, reported greater rates
of improvement in children with better residual hearing and higher
socioeconomic status. Additionally, Geers, Nicholas and Sedey
(2003), who studied 181 8;00- and 9;00-year-old children who also
received a cochlear implant by the age of five, found that the
child’s learning ability contributed greatly to the linguistic outcome. Participants with average learning ability were reported to
show language levels similar to those of their HA-matched peers.
Similar findings were reported for two Maltese children with a
cochlear implant (Tabone, 2004). However, Geers et al. (2003)
further stated that children with average or above average intelligence performed similarly to their CA-matched peers. Together,
these results show that the linguistic outcome is multifactorial.
Speech production is influenced by the child’s age at onset of
deafness, time of implantation as well as the mode of communication and rehabilitation (Robbins, 2000). CIs improve speech
perception and intelligibility (Robbins, 2006), both of which eventually improve speech production. In the two Maltese children
with CI studied by Tabone (2004), speech development was identified as delayed, implying typical developmental stages emerging with a time lag, rather than as deviant. Furthermore, the
Maltese-speaking child observed by Xuereb (2004) at 4;06 years
post-implantation experienced speech and language delay when
compared to her CA-matched peers on tests of speech and language. However, the tests used in both case studies were not
standardised on the Maltese-speaking population, so interpretations of results need to be considered as tentative.
Importantly, speech and language assessments standardised on
the Maltese hearing population have recently become available,
enabling comparative evaluation of the speech and language skills
of children with CIs in relation to those of Maltese hearing children. Such a comparison was the purpose of the current study.
The following research questions were addressed:
http://dx.medra.org/10.14614/COCHIMP.2.60
61
1. What speech and language skills are manifested by three
Maltese children with bilateral CIs?
2. Are the speech and language skills of these three Maltese children comparable to those shown by HA- or CA-matched typicallydeveloping children?
More specifically, the study documented the participants’ phonetic inventory, phonological error patterns, inconsistency in word
production, verbal comprehension and expression, aiming to identify their strengths and weaknesses. It also sought to determine
whether their skills were the same as, similar to or different from
the speech and language abilities documented in a normative sample of Maltese children. This information was needed to indicate
whether the speech and language development of the three Maltese participants with CIs was age-appropriate, delayed or deviant
in relation to hearing children, while acknowledging the limited
generalisability of results to other Maltese children with CIs.
2
2.1
Methods
Participants
All speech-language pathologists working in the public health
service were asked to distribute an information sheet among
potential participants in their current or previous caseloads.
Children considered as potential participants had bilateral
cochlear implants, a HA ranging between 2;00 and 6;00 years
and were Maltese-English bilinguals. Children reported to have
other developmental disabilities were excluded. Three children
having a HA in the range of 2;10-5;03 years were identified as
participants. Table 1 lists background information on each child.
2.2
Research design
A multiple case study design was implemented. Since this
design is known to enable researchers to investigate real-life events
holistically (Yin, 2009), its implementation in this study allowed
the chosen participants to be studied in depth. The status of
their speech and language development was assessed through
the use of standardised assessments and results were analysed
quantitatively and qualitatively. Furthermore, previous audiology
and speech and language progress reports were accessed for background information that could help in the interpretation of results.
2.3
Method
All data were collected by the first author. Background
information was obtained by reviewing the participants’ medical
and audiology file. Details on their hearing assessment results and
implantation were noted. Their HA was subsequently calculated.
Speech and language data were then collected through the use
of two standardised assessments, outlined below, which enabled
each participant’s scores to be compared with population norms.
The Maltese-English Speech Assessment (MESA) (Grech,
Dodd & Franklin, 2011) assesses speech and taps into articulation, phonology, consistency, diadochokinesis, consonantal
clusters and multisyllabic words, as well as single oral movements
and sequenced movements. In the present study, only the articulation, phonology and consistency subtests were administered.
The articulation and phonology subtests involved naming 42
coloured pictures eliciting all the English and Maltese vowels and
consonants (Grech & Dodd, 2008). This enabled compilation
of their phonetic inventory and the tabulation of phonemes.
Percentage Consonants Correct (PCC) and Percentage Vowels
Correct (PVC) scores were calculated and error patterns were
described and analysed. In the consistency subtest, the participants were to name 17 pictures, three times on separate trials.
An inconsistency score was then computed.
http://www.um.edu.mt/healthsciences/mjhs
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
Table 1. Background information on the participants chosen for the study
Child 1
Child 2
Chronological age (CA) during time of assessment (years) 4;08
6;10
Hearing age (HA) during time of assessment (years)
2;10
3;06
Type of implantation
Simultaneous
Sequential
bilateral
bilateral
Chronological age (CA) at first implant (years)
1;10
3;04
Chronological age (CA) at second implant (years)
1;10
5;10
Interval between both implants (years)
-2;06
Language acquisition*
Sequential
Sequential
bilingual
bilingual
Primary language
Maltese
Maltese
Gender
Male
Male
62
Child 3
7;06
5;03
Sequential
bilateral
2;03
3;01
0;10
Simultaneous
bilingual
English
Female
*Child 1 and Child 2 were referred to as sequential bilinguals since, in line with Iglesias and Rojas’s description (2012), they
were predominantly exposed to Maltese (L1) from an early age and then to English (L2) at school. Child 3 was referred to as
simultaneous bilingual following Genesee’s (1989, cited in Iglesias and Rojas, 2012) definition of concurrent acquisition of more
than one language during the time of primary language development. In fact, she was consistently exposed to both Maltese
and English at home, although English input was reported to be more predominant.
The Language Assessment for Maltese Children (LAMC)
(Grech, Franklin & Dodd, 2011) examines language skills, tapping into verbal comprehension, narrative vocabulary and grammar, sentence imitation, phonological awareness and pragmatics.
It also includes a voice and fluency checklist. Participants were
administered the verbal comprehension, narrative vocabulary and
grammar subtests. They were first asked questions related to the
story previously narrated by the assessor using a picture stimulus
book. They were subsequently encouraged to retell the story with
the help of the stimulus book.
Child 1 and Child 2 were assessed at their Speech and Language Clinic. Child 3, who no longer needed speech and language
therapy, was assessed at home. Child 1 and 3 completed both
assessments in one session, which took approximately 1.5 hours.
Since Child 2 was noted to be distracted following the first part
of assessment, the test battery was spread over two sessions lasting an hour each. Assessment duration was typical as during the
standardisation of MESA and LAMC, administration of each test
lasted up to 60 minutes. Assessment sessions were audio-recorded.
Information from the children’s speech and language file about
performance during previous speech and language therapy sessions
was also recorded. These details provided a holistic background
to the quantitative speech and language data collected. The participants were free to speak in their preferred language during
assessment. The primary language was taken to be the language
used spontaneously and predominantly by the child. The speech
data were transcribed phonetically while the language data were
transcribed orthographically.
2.4
Ethical considerations
Prior to commencing data collection, the study was approved by
the University of Malta Research Ethics Committee, ensuring that
the participants’ rights were safeguarded. The legal guardians of
the potential participants were given an information sheet through
their speech-language pathologist, explaining the children’s potential involvement in the study. For Child 3, the information
sheet was passed on through the speech-language pathologist previously working with the child. Upon understanding the children’s
involvement in the study and agreeing to their participation, parents completed a consent form.
http://dx.medra.org/10.14614/COCHIMP.2.60
3
Results
Descriptive statistics, including raw scores, means, standard deviations, z-scores and percentiles for each participant on every
speech and language subtest, were calculated. The children’s individual performance was compared to that of typically-developing
HA- and CA- matched peers involved in assessment standardisation. However, the available norms for both assessments cover the
3;00-6;00-year age group, with the exception of the verbal comprehension subtest, for which norms begin at 2;06 years. Grech,
Franklin and Dodd (2011) explain that children aged 2;00-2;06
years carried out only a small part of the language assessment.
The resulting data did not warrant further analysis. Consequently,
given Child 1’s actual HA of 2;10 years, he was compared to HAmatched peers aged 3;00 years. Analogously, Child 2 and Child
3, whose actual CAs were 6;10 and 7;05 years respectively, were
both compared to CA-matched peers aged 6;00 years, as this was
the ceiling age of both assessments.
When the MESA was standardised, children were divided into
monolingual and bilingual groups. As defined by Grech, Dodd
and Franklin (2011), the monolingual group consisted of children
exposed to Maltese at home and to English at school, whilst children in the bilingual group had simultaneous exposure to both
languages at home. Hence, the results of Child 1 and 2 were compared to the monolingual group norming data, whilst Child 3’s
data were compared to the bilingual group.
Each participant’s phonetic inventory was determined first. According to Ingram (1981, cited in Grech, 2006), a criterion for a
sound to become part of the child’s inventory is its frequency, in
that it has to occur at least once in a mixture of 25 words selected
at random. The phonetic inventory was noted from the articulation subtest of the MESA by listing the sounds that the child
produced. These sounds are hereafter referred to as phones. The
number of phonemes was subsequently counted from performance
on the phonology subtest. For a sound to be considered a phoneme
it should be used regularly and contrastively and it should occur
in minimal or near minimal pairs (Grech, 2006). The phonology
subtest also enabled calculation of the PCC and PVC scores. Finally, the inconsistency score was derived from the participants’
inconsistent production of words on the consistency subtest.
http://www.um.edu.mt/healthsciences/mjhs
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
63
Table 2. Child 1’s PCC, PVC and inconsistency scores (means and standard deviations (SD) quoted
from the MESA manual) with the equivalent z-scores and percentiles derived for HA- and CA-matched
peers
Raw Score Quoted
Quoted
Derived
Derived
Mean
SD
z-score
Percentile
PCC
HA: 2;10 years
91.45
89.6
9.20
0.201
58
CA: 4;08 years
91.45
97.23
2.68
-2.157
2
PVC
HA: 2;10 years
98.99
99.18
3.29
-0.058
48
CA: 4;08 years
98.99
99.89
0.35
-2.609
0.5
PVC without outlier
HA: 2;10 years
100
99.18
3.29
0.250
60
CA: 4;08 years
100
99.89
0.345
0.320
63
Inconsistency Score
HA: 2;10 years
29.41
15.05
18.59
0.748
77
CA: 4;08 years
29.41
4.03
7.79
3.258
100
Table 3. Child 1’s phonological error processes, frequency of occurrence and age
expected, based on data by Grech, Dodd and Franklin (2011)
Phonological Error Processes
Error Processes
Frequency
Systemic Error Processes
Voicing
2
Backing
2
Fronting
2
Lateralisation of /r/
2
Nasalisation
1
Structural Error Processes
Weak syllable deletion
1
Final consonant deletion
1
Initial consonant deletion
1
Compensatory vowel lengthening
1
Consonant harmony
1
Epenthesis
1
Gemination reduction
1
Cluster formation
1
at which they are
Typically expected
3;00 - 3;05 years
3;00 - 3;05 years
3;00 - 6;00 years
3;00 - 4;11 years
MEP*
3;00 - 3;05 years
3;00 - 4;11 years
3;00 - 3;11 years
MEP*
MEP*
MEP*
MEP*
MEP*
*Minimally used error pattern (MEP): an error pattern used by less than 10% of children between 3;00 and 6;00 years
3.1
Child 1
Child 1 produced a complete phone repertoire, 22 phones, which
included all the Maltese consonants documented by Azzopardi
(1981) (cited in Borg & Azzopardi-Alexander, 1997). Given the
child’s CA (4;08 years) and Grech and Dodd’s (2008) MESA results indicating that both sequential and simultaneous MalteseEnglish bilingual children produce all phones by 4;00 years, this
was an expected finding. Therefore, Table 2 shows Child 1’s raw
scores on the MESA speech subtests in comparison to his HA- and
CA-matched peers and Table 3 lists his phonological error processes.
In the light of his CA, Child 1 should have mastered the
majority of the adult phonemes (Grech, Dodd & Franklin, 2011).
The child did show a complete phonemic inventory by producing
22 phonemes. Yet, his phonological system was still developing,
with his performance similar to that of his HA-matched peers. On
PCC, he scored close to the average of his HA-matched peers yet
well below average in comparison to his CA-matched peers. A similar performance was noted on PVC. Although Child 1 performed
below average in PVC when compared to both groups, there was
a slight difference between his raw score and the mean score for
http://dx.medra.org/10.14614/COCHIMP.2.60
both age-matched groups. This was due to an omitted vowel.
Without this outlier, the child would have obtained a full score.
Also, the possibility of an inconsistent phonological disorder was
eliminated as the child obtained an inconsistency score lower than
40% (see Grech, Dodd & Franklin, 2011). However, the discrepancy between the child’s score and the mean score for both age
groups seems to suggest that he was still learning the Maltese and
English phonological systems. As discussed by Grech and Dodd
(2008), inconsistent production increases with initial exposure to
two languages.
Child 1 produced five systemic and eight structural error processes. None were considered frequently-used error patterns. Yet,
they are potential error patterns. When compared to his HAmatched group, seven error processes used by the child were all
typically used at 3;00 years. Alternatively, when compared to his
CA peers, voicing, backing, weak syllable deletion and initial consonant deletion should have been eradicated by the age of 4;08
years. Furthermore, half the error processes used by Child 1 (see
minimally used error patterns
http://www.um.edu.mt/healthsciences/mjhs
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
64
Table 4. Child 1’s scores on verbal comprehension and narrative ideas (means and standard deviations
(SD) quoted from the LAMC manual) with the equivalent z-scores and percentiles derived for HA- and
CA-matched peers
Raw Score
Quoted
Mean
HA: 2;10 years
CA: 4;08 years
11.50
11.50
10.61
16.80
HA: 2;10 years
CA: 4;08 years
14
14
0.92
9
Quoted
SD
Derived
z-score
Derived
Percentile
3.38
1.87
0.263
-2.834
60
0.2
1.44
6.14
9.083
0.814
100
79
Verbal Comprehension Score
Narrative Ideas Score
Table 5. Child 2’s PCC, PVC and inconsistency scores (means and standard deviations quoted from the
MESA manual) with the equivalent z-scores and percentiles derived for HA- and CA-matched peers
Raw Score
Quoted
Mean
HA: 3;06 years
CA: 6;10 years
96.32
96.32
93.35
95.53
HA: 3;06 years
CA: 6;10 years
98.85
98.85
99.99
100
HA: 3;06 years
CA: 6;10 years
100
100
99.99
100
HA: 3;06 years
CA: 6;10 years
35.29
35.29
10.60
3.19
Quoted
SD
Derived
z-score
Derived
Percentile
6.47
5.58
0.456
0.135
68
55
0.02
0∗
-57
-
0
-
0.02
0∗
0.5
-
69
-
14.96
6.65
1.65
4.827
95
100
PCC
PVC
PVC without outlier
Inconsistency Score
*A standard deviation of 0 indicates no variation around the mean score, so that the z-score and percentile could not be computed.
(MEP) in Table 3) were typical between 2;00 and 3;00 years,
as documented in Grech’s (1998) study. This further suggested
delayed phonological development in comparison to the child’s
CA. Child 1’s verbal comprehension and narrative ideas scores
are shown in Table 4.
Child 1 understood simple commands and ‘why’ questions.
However, comprehension of ‘who’ and ‘what’ was difficult. Therefore, his understanding of wh-questions was still emerging. As
documented by Sax and Weston (2007), typically-developing children between 2;00 and 3;00 years comprehend and produce simple wh-questions. However, Friedmann and Szterman (2011) reported that the understanding and production of wh-questions of
11 Hebrew-speaking children, some of whom were fitted with binaural hearing aids and others with a CI, was weaker compared
to typically-developing children. Child 1 also understood basic
colour terms, but knowledge of numbers and counting was still
emerging.
Child 1’s expression was mainly in Maltese. He used proper
names and target lexical nouns appropriately with correct
singular and plural forms. Various lexical target verbs and the
definite and assimilated article were also employed appropriately.
There was evidence of the use of the Maltese negative form
mhux (not), the English lexeme ‘no’ and spatial and pronominal
use of deictic expressions. On 90% of occasions, he produced
correct person and number agreement and adequate sentence
structures. On the other hand, Child 1 made limited use of
http://dx.medra.org/10.14614/COCHIMP.2.60
adjectives and adverbs, conjunctions, prepositions, direct and
indirect pronouns. He did not use possessives and showed gender
agreement difficulties. The masculine form was used for 90%
of the time. Infrequent and incorrect use of feminine forms
possibly indicated their emergence at the time. A similar trend
was documented by Coppini (2002), who investigated gender
agreement between nouns and adjectives in Maltese children.
3.2
Child 2
Child 2 produced 22 phones, which made up a complete phone
repertoire in view of his HA (3;06 years) and CA (7;10 years).
Table 5 shows his raw scores on the MESA speech subtests in
comparison to his HA- and CA-matched peers. Table 6 lists the
phonological error processes.
Child 2 produced 22 phonemes, thus he had also developed a
complete phonemic inventory. However, his phonological development was still emerging. On PCC, Child 2 performed moderately
and slightly above average compared to his HA- and CA-matched
peers respectively. Furthermore, on PVC, the slight difference
between the child’s score and that of the normative population
was mainly due to the incorrect production of /mEr5/ (mirror),
for which he said /m5r5/ (woman). Without this outlier, Child 2
obtained a full score, further indicating no vowel difficulty. His inconsistency score indicated a higher possibility of an inconsistent
http://www.um.edu.mt/healthsciences/mjhs
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
disorder.
Child 2 exhibited a chronological mismatch in the phonological
processes observed. He produced five systemic error processes
and no structural error processes. When compared to his HAmatched group, Child 2 produced developmental phonological
processes typical of children younger than his HA. Alternatively,
when compared to his CA-matched peers, backing, voicing and
stopping should have been eliminated by 3;11 years. Therefore, a
phonological delay was observed. Conversely, as expected by his
CA, Child 2 did not exhibit structural error processes. Thus, he
followed some patterns expected at 6;10 years.
Child 2’s verbal comprehension and narrative ideas scores are
shown in Table 7.
Child 2 understood simple commands and ‘why’ questions.
Comprehension of ‘where’ and ‘what’ questions, as well as
knowledge of numbers and counting was emerging. He made
65
appropriate regular lexical choices of nouns and produced the
Maltese negative form ma (verb)x. He made regular and adequate
use of articles and conjunctions. On 90% of occasions, he also
made correct verb choices and used adequate pronouns. The
direct object was used appropriately but infrequently, on less
than 30% of occasions. Child 2 showed correct gender, number
and person agreement. On the other hand, he replaced proper
names with nouns and made adequate use of prepositions while
omitting others. His use of adjectives and adverbs amounted to
less than 30% of their required usage. He rarely employed spatial
deictic expressions and sentence structure was occasionally
disrupted. The indirect object was incorrectly used due to person
and number disagreement and possessive terms did not emerge.
Table 6. Child 2’s phonological error processes, frequency of occurrence and age at which they are
expected based on data by Grech, Dodd and Franklin (2011)
Phonological Error Processes
Error Processes
Frequency Typically expected
Systemic Error Processes
Backing
3
3;00 - 3;05 years
Devoicing
1
3;00 - 3;05 years
Stopping
1
3;00 - 3;11 years
Delteralisation of /l/
1
MEP*
Frictation
1
MEP*
*Minimally used error pattern (MEP): an error pattern used by less than 10% of children between 3;00 and 6;00 years
Table 7. Child 2’s scores on verbal comprehension and narrative ideas (means and standard deviations
(SD) quoted from the LAMC manual) with the equivalent z-scores and percentiles derived for HA- and
CA-matched peers
Raw Score Quoted
Quoted
Derived
Derived
Mean
SD
z-score
Percentile
Verbal Comprehension Score
HA: 3;06 years
10.5
14.87
2.50
-1.748
4
CA: 6;10 years
10.5
17.21
2.43
-2.761
0.2
Narrative Ideas Score
HA: 3;06 years
16
2.88
3.19
4.113
100
CA: 6;10 years
16
16.86
6.30
-0.137
45
Table 8.Child 3’s PCC, PVC and inconsistency scores (means and standard deviations quoted from the
MESA manual) with the equivalent z-scores and percentiles derived for HA- and CA-matched peers
Raw Score Quoted
Quoted
Derived
Derived
Mean
SD
z-score
Percentile
PCC
HA: 5;03 years
100
99.34
2.21
0.299
62
CA: 7;06 years
100
99.18
1.24
0.661
75
PVC
HA: 5;03 years
100
99.94
0.21
0.280
61
CA: 7;06 years
100
99.74
0.83
0.313
62
Inconsistency Score
HA: 5;03 years
5.88
0.45
1.63
3.331
100
CA: 7;06 years
5.88
1.32
2.62
1.740
96
http://dx.medra.org/10.14614/COCHIMP.2.60
http://www.um.edu.mt/healthsciences/mjhs
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
66
Table 9. Child 3’s verbal comprehension and narrative ideas scores (means and standard deviations
(SD) quoted from the LAMC manual) with the equivalent z-scores and percentiles derived for HA- and
CA-matched peers
Raw Score Quoted
Quoted
Derived
Derived
Mean
SD
z-score
Percentile
Verbal Comprehension Score
HA: 5;03 years
18.5
19
1
-0.500
31
CA: 7;06 years
18.5
18
1.41
0.355
64
Narrative Ideas Score
HA: 5;03 years
35
29.33
4.04
1.403
92
CA: 7;06 years
35
23
7.07
1.697
96
3.3
Child 3
Child 3 produced 23 phones. As expected for her HA (5;03
years) and CA (7;05 years), Child 3 produced all phones reported
by Azzopardi (1981) (cited in Borg & Azzopardi-Alexander, 1997)
including the voiced velar nasal [ŋ] which pertains to English
phonology. Table 8 shows her raw scores on the MESA speech
subtests in comparison to her HA- and CA-matched peers.
Child 3 produced 23 phonemes, thus showing availability of a
complete phonemic inventory. She obtained full score in PCC and
PVC. When compared to her age-matched groups, she performed
above average. Additionally, Child 3 exhibited no phonological
error patterns.
Table 9 shows Child 3’s verbal comprehension and narrative
scores. Just like typically-developing children, Child 3 was able
to attend to the story while replying correctly to the questions
asked. She understood simple commands, basic colour terms and
why-questions. She had mastered the knowledge of numbers and
counting. Her responses to questions during the comprehension
subtest showed evidence of verbal reasoning. Yet, she did not understand the spatial term ‘on’. Child 3 made good lexical choices
of nouns, verbs, adjectives and adverbs. She produced the negative ‘not’, together with lengthy and informative sentences. Conjunctions, prepositions, pronouns, articles, possessive determiners,
as well as gender, person and number agreement, were adequately
used. Conversely, no deictic terms were employed by the child.
4
Discussion
The aim of this study was to describe and evaluate the speech
and language skills of three children with bilateral CIs, to derive information on their strengths and weaknesses in relation to
their HA- and CA- matched peers. The participants were assessed
using three speech and two language subtests of the MESA and
LAMC respectively, all of which were standardised on the Maltese
population. Discussion of results follows and acknowledges the
potential influence of each child’s internal characteristics, learning environment and speech and language therapy on their speech
and language performance.
Child 1 followed typical speech development. Yet, his scores
indicated that he was on a par with his HA group whilst delayed when compared to his CA-matched peers. This was consistent with Tabone’s (2004) and Xuereb’s (2004) conclusions on
the speech development of Maltese children with CI. On the verbal comprehension subtest, Child 1 performed within the average
range compared to his HA-matched peers. Caselli et al. (2012)
reported similar trends in lexical comprehension for 17 Italian
children who received a CI at 2;00 years of age.
The child’s generation of expressive ideas on narrative was excellent when compared to his HA-matched peers. Performance was
above average when compared to his CA-matched peers. Therefore, at 2;10 years post-simultaneous bilateral implantation, the
child’s expressive skills were progressing well. This concurred with
http://dx.medra.org/10.14614/COCHIMP.2.60
Wie’s (2010) findings that the majority of children simultaneously
implanted acquire both receptive and expressive language skills
at a faster pace than expected in comparison to their HA. As observed from the reviewed files, the family members provided good
support to the child. This was well reflected in his expressive language, which was developing at a steady pace. Additionally, it was
reported by his speech-language pathologist that co-operation,
motivation and carryover of therapy in the natural environment
contributed to the child’s positive prognosis. Furthermore, the
fact that the child was given a shared learning support assistant
(LSA) from kindergarten years enhanced and supported his educational needs. The speech-language pathologist liaised with the
child’s LSA to consolidate the child’s learning in both the school
and clinical contexts.
Child 2’s speech development followed the pattern of typicallydeveloping children. Nevertheless, his scores were similar to his
HA-matched peers and sometimes even slightly delayed in comparison. He exhibited typical phonological error processes with a
chronological mismatch, since he used error processes from different age groups with inconsistent production of words. The latter
might be indicative of incomplete knowledge of two phonological systems. It seems reasonable to associate high variability in
speech production with children having the least hearing experience. However, this was still evident at the HA of 3;06 years,
reflecting instability in adult-like word production.
Child 2’s performance on verbal comprehension was considerably poor when compared to both age groups. His difficulty understanding wh-questions was possibly due to the delayed auditory
stimulation of 3;04 years. In fact, his audiology file reported that
his left ear, being the worse one, was implanted at 3;04 years while
he was wearing the right hearing aid on a full-time basis. Yet, as
documented in his audiology file, Child 2 seldom made use of this
hearing aid. He received his second implant at 5;10 years. Scherf
et al. (2009) observed improved auditory skills even in children
receiving their second CI after the age of 6;00 years. Yet, older
children obtained comparable results two years following bilateral
use. In the case of Child 2, bilateral use had been in effect for one
year. This suggests that further bilateral experience was required.
In contrast, Child 2’s narrative ideas were excellent when compared to his HA-matched group and slightly below average when
compared to his CA-matched peers. This discrepancy between
his receptive and expressive skills is not what is normally experienced by a typically- developing child. The wh-questions were
likely to have impeded his performance on the comprehension subtest. From the assessments carried out by his speech-language
pathologist, the child showed understanding of commands with
two to three information carrying words. Therefore, rewording of
the comprehension assessment questions and substitution of words
like ‘bniet’ (girls) with simpler ones might have possibly enhanced
his performance. However, since both assessments used are standardised, this was not possible.
Additionally, Child 2 had few instances of unintelligibility which
http://www.um.edu.mt/healthsciences/mjhs
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
might have been due to the manifestation of systemic error processes. Allen, Nikolopoulos and O’Donoghue (1998) concluded
that children with CI continue to develop intelligibility even after
five years of implantation. Also, during the speech and language
therapy sessions following implantation, emphasis was
R placed on
the production of Ling sounds (/5/, /0/, /i/, /m/, / /, /s/) and
thus, not all the speech sounds were targeted. This might have
also possibly contributed to unintelligibility in the production of
words. As in the case of Child 3, speech and language therapy
targeted all the speech sounds in all the different word positions
and also within sentences.
Child 3 showed typical speech skills. Her performance was similar to her CA-matched peers. Yet, unexpectedly, Child 3 obtained a higher percentile when compared to her CA than when
compared to her HA-matched group. However, Grech and Dodd
(2007) noted a dip in the phonological developmental profile of
5;00-5;05-year-old typically-developing bilingual children (Child
3’s HA-matched group). Thus, the child’s performance was typical for her HA. Moreover, she showed adequate phonological proficiency in both Maltese and English. This could have been due
to her early exposure to both languages.
The language subtests also showed Child 3 to perform better in
comparison to her CA- than to her HA-matched group. This led
to the reliability of the derived equivalent z-scores and percentiles
being queried. As discussed in the LAMC manual, z-score accuracy may be low when used for smaller samples (Grech, Franklin
& Dodd, 2011). The English norming group to whom Child 3 was
compared was small (N = 30) compared to the Maltese group (N
= 205).
Child 3 performed well above average compared to both groups
of age-matched peers. She had outgrown her HA peers and performed closely to her CA-matched peers. Her hearing experience
of 5;03 years might have influenced her skills, given that she had
the longest exposure to language when compared to the other participants in this study. In fact, Schramm, Bohnert and Keilmann
(2010) concluded that children with CIs increase their linguistic
abilities with their HA, that is, the longer the child is fitted with
cochlear implants, the better the linguistic outcome. Furthermore,
Child 3 was the only female participant in the study. Schramm et
al. (2010) observed that better language competence was developed by females, possibly indicating that this factor might have
played a role in her linguistic performance.
Interestingly, and unlike the other two participants, it was revealed by Child 3’s file that following the diagnosis of hearing loss,
she attended sessions with a teacher of the hearing-impaired, during which she was taught baby signs. It was reported that the child
immediately picked up the signs and used them communicatively.
Having the means to communicate from an early age might have
therefore limited the gap in relation to her CA-matched peers.
Also, following the implantations, Child 3 attended speech and
language therapy sessions on a weekly basis. At the time of testing, the child did not attend therapy as speech and language difficulties were no longer reported. Furthermore, although Child
3 performed similarly to her CA-matched peers (6;00 years), her
actual CA was 7;06 years. Thus, one needs to be cautious when
interpreting her results, as a fair and accurate comparison would
have ideally involved her actual CA.
It is acknowledged that this study presented with shortcomings
which could not be controlled. These called for cautious interpretation of results. Data on Maltese children with bilateral CIs were
not available. Thus, reference was made to sparse data on Maltese children with unilateral CI and comparison was then made
to results of foreign research studies. However, exact comparisons could not be carried out due to the different languages and
methodologies used. Data on Maltese typically-developing children were also limited. Moreover, generalisation of the results
was not possible due to the case study design and the individual
variability across the participants, which might have contributed
http://dx.medra.org/10.14614/COCHIMP.2.60
67
to the differences in scores. Therefore, each participant was analysed and discussed separately.
5
Conclusion
The results of the current study shed light on the speech and
language performance of three Maltese children with bilateral
CIs. These findings are preliminary and cannot be generalised.
However, with respect to the three participants, it appears that
cochlear implantation provides relevant auditory information that
helps develop oral communication. It was concluded that two of
the participants presented with delayed but typical speech development, whilst the third child performed on a par with her CAmatched peers. With increased CI experience, phonetic accuracy
improved. Additionally, the expressive language score was higher
than verbal comprehension for all participants. It emerged that
the earlier the cochlear implantation, the shorter was the gap in
sequential implants. Moreover, better speech and language outcomes were observed with increased experience with CIs. Results
showed that early cochlear implantation enabled the participants
to engage in a typical school environment with their HA-matched
peers.
6
Acknowledgements
The authors are grateful to Professor Helen Grech for granting them permission to use the standardised assessments and
their respective norms, and to the audiologist and speech-language
pathologists who helped in identifying the participants. Warmest
thanks go to the parents of the participants for consenting to their
children’s participation and to the children themselves, who patiently and cheerfully co-operated during assessment.
7
Funding
This research has received no specific grant from any funding
agency in the public, commercial or non-profit sectors.
8
Conflicts of Interest
The authors report no conflicts of interest.
References
Allen, M.C., Nikolopoulos, T.P. & O’Donoghue, G.M. (1998)
Speech intelligibility in children after cochlear implantation.
The American Journal of Otology, 19(6), pp. 742-746.
Archbold, S. (2002) Cochlear implants. In W. McCracken & S.
Laoide-Kemp (Eds) Audiology in Education (pp. 239-266).
London: Whurr Publishers.
Battista, R. A. & Highhouse, K. (2011) Bilateral cochlear implants. Audiologyonline, Article #840. [Online] Available
from: www.audiologyonline.com. [Accessed 5th September
2013].
Borg, A. & Azzopardi-Alexander, M. (1997) Maltese. London:
Routledge.
Caselli, M. C., Rinaldi, P., Varuzza, C., Giuliani, A. & Burdo, S.
(2012) Cochlear implant in the second year of life: lexical
and grammatical outcomes. Journal of Speech, Language,
and Hearing Research, 55, pp. 382-394. doi:10.1044/10924388(2011/10-0248).
Coppini, A. (2002) The acquisition of number and gender agreement between nouns and adjectives in 3 to 5 year old
Maltese-speaking children. Unpublished B.Sc. (Hons) dissertation. Malta: University of Malta.
Dunn, C.C., Tyler, R.S., Oakley, S. & Gantz, B.J. (2008) Comparison of speech recognition and localization performance
in bilateral and unilateral cochlear implant users matched
http://www.um.edu.mt/healthsciences/mjhs
Speech and language skills of Maltese children with bilateral cochlear implants: three case studies
on duration of deafness and age at implantations. Ear and
Hearing, 29(3), pp. 352-359.
Friedmann, N. & Szterman, R. (2011) The comprehension and
production of WH-questions in deaf and hard-of-hearing
children. Journal of Deaf Studies and Deaf Education,
16(2), pp. 212-235.
Geers, A. E., Nicholas, J. G. & Sedey, A. L. (2003) Language
skills of children with early cochlear implantation. Ear and
Hearing, 24(1), pp. 46S-58S.
Grech, H. (1998) Phonological development of normal Maltese
speaking children. Unpublished Ph.D. dissertation. United
Kingdom: University of Manchester.
Grech, H. (2006) Phonological development of Maltese-speaking
children. In Z. Hua & B. Dodd (Eds) Phonological Development and Disorders in Children: A multilingual perspective
(pp. 64-73). Clevedon: Multilingual Matters.
Grech, H. & Dodd, B. (2007) Effects of language learning context
on phonological acquisition. Paper presented at the First
International Conference of Maltese Linguistics. Germany:
Bremen University.
Grech, H. & Dodd, B. (2008) Phonological acquisition in Malta: a
bilingual language learning context. International Journal
of Bilingualism, 12(3), pp. 155-171.
Grech, H., Dodd, B. & Franklin, S. (2011) Maltese-English Speech
Assessment (MESA). Malta: University of Malta.
Grech, H., Franklin, S. & Dodd, B. (2011) Language Assessment
for Maltese Children (LAMC). Malta: University of Malta.
Iglesias, A. & Rojas, R. (2012) Bilingual language development
of English language learners: modelling the growth of two
languages. In B. A. Goldstein (Ed) Bilingual Language Development and Disorders in Spanish-English Speakers (2nd
ed.) (pp. 3-30). Baltimore: Paul H. Brookes.
Kim, J-S., Kim, L-S. & Jeong, S-W. (2013) Functional benefits of
sequential bilateral cochlear implantation in children with
long inter-stage interval between two implants. International Journal of Pediatric Otorhinolaryngology, 77, pp.
162-169.
Litovsky, R., Parkinson, A., Arcaroli, J. & Sammeth, C. (2006)
Simultaneous bilateral cochlear implantation in adults: a
multicenter clinical study. Ear and Hearing, 27(6), pp. 714731.
Niparko, J. K., Tobey, E. A., Thal, D. J., Eisenberg, L. S., Wang,
N. Y., Quittner, A. L. & Fink, N. E. (2010) Spoken language development in children following cochlear implantation. The Journal of the American Medical Association
(JAMA), 303(15), pp. 1498-1506.
Robbins, A. M. (2000) Rehabilitation after cochlear implantation.
In J. K. Niparko (Ed.) Cochlear Implants: Principles and
practices (pp. 323-363). Philadelphia: Lippincott Williams
& Wilkins.
Robbins, A. M. (2006) Language development in children with
cochlear implants. In S. B. Waltzman & J. T. Roland (Eds)
Cochlear Implants (2nd ed.) (pp. 153-166). New York:
Thieme Medical Publishers.
Roland, J. T. (2000). Complications of cochlear implant surgery.
In S. B. Waltzman & N. L. Cohen (Eds) Cochlear Implants
(pp. 171-175). New York: Thieme Medical Publishers.
Sammeth, C. A. (2007) Why two is better than one - the benefits of
bilateral cochlear implants. Implantable Hearing Solutions,
1(3), pp. 14 - 16.
Sax, N. & Weston, E. (2007) Language development milestones.
[Online] Available from: (http://www.rehabmed.ualberta.
ca/spa/phonology/milestones.pdf [Accessed 20th August
2014].
Scherf, F., van Deun, L., van Wieringen, A., Wouters, J., Desloovere, C., Dhooge, I., Offeciers, E., Deggouj, N., De Raeve,
L., Wuyts, F. & van de Heyning, P. (2009) Three-year
postimplantation auditory outcomes in children with se-
http://dx.medra.org/10.14614/COCHIMP.2.60
68
quential bilateral cochlear implantation. Annals of Otology,
Rhinology & Laryngology, 118(5), pp. 336-344.
Schramm, B., Bohnert, A. & Keilmann, A. (2010) Auditory,
speech and language development in young children with
cochlear implants compared with children with normal hearing. International Journal of Otorhinolaryngology, 74(7),
pp. 812-819. doi:10.1016/j.ijporl.2010.04.008.
Sharma, A., Gilley, P. M., Martin, K., Roland, P., Bauer, P. &
Dorman, M. (2007) Simultaneous versus sequential bilateral
implantation in young children: effects on central auditory
system development and plasticity. Audiological Medicine,
5, pp. 218-223.
Tabone, N. (2004) Four case studies on the language skills of
hearing-impaired children with hearing aids and cochlear
implants. Unpublished B.Sc. (Hons) dissertation. Malta:
University of Malta.
Therres, M. (2012) Therapy: same or different for a child with two
cochlear implants? Audiologyonline, Article #7027. [Online] Available from: www.audiologyonline.com. [Accessed
20th August 2014].
Welling, D. R. (2010) Hearing and language development. In B.
B. Shulman & N. C. Capone (Eds) Language Development:
Foundations, processes, and clinical applications (pp. 95134). U.K.: Jones and Bartlett Publishers.
Wie, O. B. (2010) Language development in children after receiving bilateral cochlear implants between 5 and 18 months.
International Journal of Pediatric Otorhinolaryngology,
74(11), pp. 1258-1266.
Xuereb, J. (2004) Language skills in a cochlear-implanted child: a
case study. Unpublished B.Sc (Hons) dissertation. Malta:
University of Malta.
Yin, R. K. (2009) Case Study Research: Design and methods (4th
ed.). London: U.K.: Sage Publications.
http://www.um.edu.mt/healthsciences/mjhs
Malta Journal of Health Sciences
http://www.um.edu.mt/healthsciences/mjhs
Table of Contents
Malta Journal of Health Sciences Volume 1 - Issue 2 (December 2014)
Editorial
28
Editorial
Daniela Gatt
Regular Articles
29
Key environmental stress biomarker candidates for the optimisation of
chemotherapy treatment of leukaemia Review Article
Eirini G. Velliou, Susana Brito Dos Santos, Maria Fuentes-Garí, Ruth
Misener, Eleni Pefani, Nicki Panoskaltsis, Athanasios Mantalaris and
Efstratios N. Pistikopoulos
35
Degree of burnout among Maltese podiatrists working in two different
settings Research Article
Trista Agius and Cynthia Formosa
40
The effectiveness of social communication groups in mainstream schools
Research Article
Tiziana Baldacchino and Kristina Agius
54
The lived experience of persons with an amputation Research Article
Chanelle Grech and Roberta Farrugia Debono
60
Speech and language skills of Maltese children with bilateral cochlear
implants: three case studies Research Article
Martina Galdes and Daniela Gatt