Management and Diagnosis of Psycogenic Cough, Habit Cough, and Tic Cough: A Systematic Review

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Management and Diagnosis of Psycogenic Cough, Habit Cough, and Tic Cough: A

Systematic Review

Qusay Haydour, MD,1 Fares Alahdab,1 Magdoleen Farah,1 Patricia Barrionuevo Moreno,1 Anne

E. Vertigan,2 Peter A. Newcombe,3 Tamara Pringsheim,4 Anne Bernadette Chang,5 Bruce K.

Rubin,6 Lorcan McGarvey,7 Kelly Ann Weir,8 Kenneth W. Altman,9 Anthony Feinstein,10

Mohammad Murad,1 Richard S. Irwin11

1Mayo Clinic, The Knowledge and Evaluation Research Unit and the Center for the Science of Health Care

Delivery, Rochester, MN

2John Hunter Hospital, Department of Speech Pathology, Newcastle, New South Wales, Australia

3University of Queensland, School of Psychology, Brisbane, Australia

4University of Calgary, Calgary, Alberta, Cananda

5Royal Children’s Hospital and Menzies School of Health Research, Charles Darwin University, Brisbane, Brisbane,

Queensland, Australia

6Virginia Commonwealth University, Richmond, VA

7The Queen’s University of Belfast, Department of Medicine, Belfast, United Kingdom

8University of Queensland, Paediatrics and Child Health, Brisbane, Queensland, Australia

9Mount Sinai Hospital, New York, NY

10Sunnybrook Health Sciences Centre, Toronto, Canada

11UMass Memorial Medical Center, Worcester, MA

Corresponding Author: Qusay Haydour, Mayo Clinic, Rochester, MN;

[email protected]

Conflicts of Interest: Dr. Kenneth Altman discloses that he has no conflict of interest in the last 22 months. In the last 3 years, Dr. Kenneth Altman had the following conflicts: Nestle (consultant), last 7/2012 and Watermark Research Partners (for Stryker), last 5/2012. Because Dr. Richard Irwin is the Editor in Chief of the Journal as well as an author on this article, he reports that he did not participate in the Journal’s review and decision process of the article. All other authors declare no conflict of interest.


mailto:[email protected]

2

Abstract:

Background: Several pharmacological and non-pharmacological therapeutic options have been

used to treat cough that is not associated with a pulmonary or extrapulmonary etiology.

Methods: We conducted a systematic review to summarize the evidence supporting different

cough management options in adults and children with psychogenic, tic and habit cough.

Medline, EMBASE, the Cochrane Central Register of Controlled Trials, Cochrane Database of

Systematic Reviews and Scopus were searched from the earliest inception of each database to

September 2013. Content experts were contacted and we searched bibliographies of included

studies to identify additional references. Results: A total of 18 uncontrolled studies were

identified enrolling 223 patients (46% males, 96% children and adolescent). Psychogenic cough

was the most common descriptive term used (90% of the studies). 95% of the patients had no

cough during sleep; barking or honking quality of cough was described in only 8 studies.

Hypnosis (3 studies), suggestion therapy (4 studies), and counseling and reassurance (7 studies)

were the most commonly used interventions. Hypnosis was effective in resolving cough in 78%

of the patients and improving it in another 5%. Suggestion therapy resolved cough successfully

in 96% of the patients. The greatest majority of improvements noted with these forms of therapy

occurred in the pediatric age group. The quality of evidence is low due to the lack of control

groups, the retrospective nature of all the studies, heterogeneity of definitions and diagnostic

criteria, and the high likelihood of reporting bias. Conclusion: Only low quality evidence

exists to support a particular strategy to define and treat psychogenic, habit and tic cough. Patient

values, preferences, and availability of potential therapies should guide treatment choice.

Introduction:

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Cough is one of the most frequently encountered symptoms in medical practice. It plays an

essential role in clearing the airway of secretions and foreign bodies; however, it can pose huge

social and economic burdens on some patients 1-4

. Chronic cough, persisting beyond 4 weeks in

children and 8 weeks in adults, can become protracted where it can cause anxiety and social

discomfort. It can be the only presenting symptom in many pulmonary and extrapulmonary

conditions. Chronic cough has to be approached systematically to better evaluate and reach the

right diagnosis 5,6

. Upper airway cough syndrome (previously referred to as postnasal drip

syndrome), asthma, nonasthmatic eosinophilic bronchitis and gastroesophageal reflux disease are

the most common causes of chronic cough in adults 7. However, an etiology of cough is not

always easily identified even after a thorough systematic investigation and psychological and

neurological conditions are in the differential diagnosis in both children and adults 8.

Various terms including habit cough, psychogenic cough and tic cough were used to describe

cases without a clear pulmonary or extrapulmonary (e.g., gastroesophageal reflux disease)

etiology in the presence of some suggestive clinical characteristics and/or an association with

psychological issue. However, these terms are not clearly defined or distinguished from each

other. The literature suggests that the classic features of these conditions include repetitive loud

dry honking cough and absence of cough during night time 9-12

. Holinger found that psychogenic

cough was the second most common cause of chronic cough in children of age 6-16 years 13

.

Many interventions including pharmacotherapy, behavioral modifications and psychotherapy

were used but none have been rigorously evaluated 9-11,14

. The concept of psychogenic cough and

its related disorders as reviewed here is distinct from other presumed etiologies of the

unexplained cough, such as “neurogenic cough” or post-viral vagal neuropathy (which is

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considered more strictly to be a disorder of the peripheral receptors or brainstem reflex feedback

loop).

The American College of Chest Physicians (ACCP) develops clinical practice guidelines to help

patients and physicians in decision-making. To aid the development of guidelines for these

challenging conditions, we conducted a systematic review of the available literature on the

management of habit cough, psychogenic cough and tic cough. Knowing of the paucity of

controlled trials and the heterogeneity of diagnostic terms, we included all study designs and

descriptive diagnostic terms.

Methods:

This systematic review follows an a priori established protocol developed by the guideline

methodologist and selected members of the ACCP expert cough panel. This report was written

following the preferred reporting items for systematic reviews and meta-analyses: the PRISMA

statement 15

.

Eligibility criteria

We searched for studies of any study design that enrolled children or adults with psychogenic

cough, habit cough, and tic cough. We included studies regardless of their language or

publication status. Case-series with two patients or more were included. Single case reports were

excluded.

Study identification

We conducted a comprehensive search of several databases from each database’s earliest

inception to September 2013. The databases included Ovid Medline In-Process & Other Non-

Indexed Citations, Ovid MEDLINE, Ovid EMBASE, Ovid PsycInfo, Ovid Cochrane Central

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Register of Controlled Trials, Ovid Cochrane Database of Systematic Reviews, and Scopus. The

search strategy was designed and conducted by an experienced librarian with input from the

guideline methodologist and selected members of the ACCP expert panel. The search used both

controlled vocabulary and keywords. The strategy used is described in the appendix. The

reference lists from the narrative reviews and existing guidelines 16-18

were searched and

consultation with experts in the field was performed to obtain any additional references that

might have been missed by the electronic search strategy.

Reviewers working independently and in duplicate reviewed all abstracts. Upon retrieval of

potentially eligible studies, the full text publications were evaluated for eligibility. The chance-

adjusted inter-reviewer agreement was calculated using the κ statistic for full text screening (κ

=0.80). Disagreements were resolved by a third reviewer.

Quality assessment

Reviewers working independently and in duplicate analyzed the full text of eligible articles to

assess the reported quality of the methods. Since all the included studies were case-series, a

modified Newcastle-Ottawa scale was used 19

. We assessed the following four elements:

selection of patient, percent lost to follow-up, ascertainment of outcome and length of follow-up.

Data extraction

Reviewers working independently and in duplicate used a standardized web-based form to

extract for each eligible study the following data items: study design, study population, study

main objective, number of patients; age and gender, number of children, description of cough

(duration, frequency, severity, triggers, cough during sleep, and associated symptoms), validated

cough assessment tools, impact on school or job, failure of conventional treatments, length of

follow-up, and number of patients who improved/failed the treatment.

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Results:

Characteristics of included studies:

Study selection process is depicted in Figure1. This evidence summary included 223 patients

enrolled in 18 uncontrolled studies published between 1966 and 2007. 81 were males (46% of

patients where gender was reported). 13 studies enrolled children and adolescents only and 2

studies enrolled mainly adults. Of the 170 patients in whom age was reported, 96% were children

and adolescents. All the included studies were retrospective case-series or case-reports. More

than half of the 223 patients were reported in 3 larger case-series: Anbar 2004, Bhatia 2002 and

Cohlan 1984 11,20,21

. The characteristics of included studies are summarized in Table 1. The

description of cough is available in Table 2. The methodological quality indicators of the studies

are in Table 3. In general, the studies were uncontrolled and considered to be at high risk of bias.

Cough characteristics:

Psychogenic cough was the term used in more than 90% of the studies either exclusively or with

other terms. Six studies used the terms habit and psychogenic interchangeably. Tic cough was

not used exclusively in any study, yet the term “psychogenic cough tic” was mentioned in three

studies. None of patients had any motor tics other than those affecting the larynx; however, one

study reported twin patients with Tourette syndrome who had phonic tic manifested as cough tic

22. None of the studies used any validated cough assessment tool. There was no clear diagnostic

criteria followed in any of the studies other than describing the symptoms with which the

patients presented. Nonproductive explosive barking/honking cough were the most common

reported cough features but these characteristics were only reported in 8 of the studies. 5%

(7/147 patients for whom this information was reported) of patients had cough during sleep.

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Duration of cough varied widely among patients with a range of two weeks to 16 years. About

half of the studies reported triggers and precipitating factors of which upper respiratory

infections and social distress were the most predominant (Table 2).

Four studies (Anbar 2004, Bhatia 2002, Blager 1988 and Bordoy 2004 11,20,23,24

) reported that

some of their patients were diagnosed with comorbid psychiatric disorders. The diagnosis was

obtained through formal psychiatric evaluation in three of the studies. Bhatia and colleagues

reported that 20 patients (62% of total patients) had comorbid psychiatric disorders which were

diagnosed according to ICD-10; the 3 most common were conversion disorder (22%), mixed

anxiety and depressive disorder (12%) and generalized anxiety disorder (10%) 11

. Anbar and

colleagues reported that conversion disorder was diagnosed in 6 patients (11% of total patients)

and anxiety disorder in 1 patient (2%) 20

. Blager and colleagues reported that 3 patients (75%)

met the criteria for the DSM-3 of conversion disorder and one patient met the criteria for

somatization disorder 23

. Bordoy and colleagues reported that all 6 patients had generalized

anxiety disorder 24

. The social and personal burdens of this cough on children were reported in 8

studies where more than 69 patients missed a period of school attendance ranging from a few

days to 6 months. The professional and social lives of 4 adult patients were also affected and

disturbed.

Interventions:

Fourteen studies reported using conventional pharmacological interventions (mostly in children)

prior to the diagnosis of psychogenic cough including antibiotics, cough suppressants,

antihistamines, bronchodilators and steroids (Table 2). However, most of these interventions

were briefly discussed and no estimate of effects was reported. In general, studies reported lack

of benefit of pharmacological interventions and focused on non-pharmacological interventions.

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In terms of antipsychotic medications, Tan and colleagues reported two boys (twin) with chronic

cough which turned out to be phonic tic of Tourette’s syndrome 22

. One of the boys was

successfully treated with haloperidol whereas cough spontaneously resolved in the other boy.

Non-pharmacological interventions were reported in most studies (Table 4). The effect of these

approaches in the different studies is summarized in Table 5. Hypnosis, suggestion therapy,

counseling and reassurance were the most commonly reported interventions.

Hypnotherapy/self-hypnosis

Hypnosis was evaluated in 96 patients who received the hypnosis instructions from a

pulmonologist or psychologist trained in hypnosis. Cough resolved in 78% and improved in 5%

of the patients.

Suggestion therapy

Four studies utilized suggestion therapy and two of them also used either a bed sheet wrapped

around the patient’s chest or nebulized lidocaine diluted in normal saline as a distractor. Cough

resolved in 96% of the 52 patients who received the intervention.

Reassurance, counseling, referral to psychologist and appropriate medications

Seven studies have used a mix of interventions including reassurance, counseling, relaxation

techniques, referral to psychologist, psychotherapy and appropriate medications (tranquilizers,

anxiolytic, and antidepressants). 93% of patients who received these interventions showed

improvement in cough.

Vocal fold injection with botulinum toxin type A

Sipp and colleagues reported treating 3 cases of debilitating cough with vocal fold injection of

botulinum toxin type A 25

. The treatment was reported as effective in breaking the cough cycle in

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all three children. However, the cough recurred after a period of time ranged from 10 days to 2.5

month and behavioral therapy was successful in controlling the cough afterwards.

Voice therapy techniques and psychotherapy

Blager and colleagues showed that voice therapy techniques and psychotherapy were effective in

improving cough in three out the four adult patients who received the intervention 23

.

Cognitive psychotherapy

Cognitive psychotherapy was reported in one study to be an effective treatment for two cases of

psychogenic cough.

Discussion:

We conducted a systematic review to summarize the evidence supporting the different treatment

strategies for psychogenic, habit and tic cough. We found 18 studies that involved 223 patients.

Different terms were used in the literature to describe the cough; 10 terms were reported in one

study 11

. We found low quality evidence supporting all the strategies used in the studies because

of the uncontrolled (non-comparative) nature of each of the studies 26

. In addition, we consider

the presence of reporting bias likely to be very high. In general, pharmacological interventions

were reported to be ineffective. Several non-pharmacological strategies were reported to be

effective.

Suggestion therapy techniques essentially referred to the process of the physician convincing the

patient that he/she can control the cough by resisting the urge to cough. The physician explains

the nature of the cough to the patient and his/her family and expresses confidence in the patients’

ability to stop the cough. Distractors of a bed sheet wrapped around the patient’s chest or

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nebulized lidocaine diluted in normal saline have been used during the process. The role of the

distractor is to help the patients control their cough and that should be explained clearly to the

patients in a way that corresponds to the nature of the cough 14

.

Despite the lack of well-defined diagnostic criteria, the literature has suggested that cases of

psychogenic, habit or tic cough have some common clinical characteristics that predominant in

many of the reported cases. It has been previously reported that a barking or honking quality of

cough and absence of cough at night are suggestive of psychogenic, habit and tic cough 27

.

However, barking or honking cough was only present in 8 of the studies and nighttime coughing

was reported in 3 studies in this review. A prospective study in adults has showed that the

absence of cough during sleep is not diagnostically helpful because other etiologies (e.g.,

chronic bronchitis and emphysema) can present with the same feature 28

. Moreover, in another

prospective study in adults, honking and barking characteristics were also found not to be useful

in diagnosing the cause of cough 29

. Given the lack of both specific clinical features and

diagnostic tests, cases of psychogenic cough have become essentially a diagnosis of exclusion 3.

Hence, diagnostic tests have been used to rule out other possible, more common, etiologies of

cough before making a diagnosis of psychogenic, tic or habit cough.

Of note is that the word ‘psychogenic’ does not appear as a descriptor in any of the DSM-5

diagnostic categories used to define physical symptoms which are incompatible with recognized

neurological or medical conditions. Furthermore, the word ‘organic’ was deleted from the DSM

taxonomy in 1994 reflecting a consensus that all mental phenomena arise from a disturbance in

brain function. Changing semantics, however, is a lot easier than changing traditions and clinical

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practices. Both terms linger prominently as the current literature relating to cough reveals. The

DSM-5 categories that capture what has been termed psychogenic cough are either somatic

symptom disorder or conversion disorder. The term conversion applies only in the context of

altered neurological function, so if cough is viewed in this light, this would be the more correct

of the two nomenclatures 30,31

. Similarly, the term “habit disorders” which previously was used

to refer to tic disorders, trichotillomania, complex motor stereotypies and skin picking, is no

longer used in the DSM. By DSM nosology, an isolated and persistent tic cough or habit cough

would be considered a chronic vocal tic disorder.

If one considers tic cough in the setting of chronic tic disorders such as Tourette’s Disorder or

Chronic Vocal Tic Disorder, there is evidence supporting both pharmacological therapy and

behavioral therapy for tics. Coughing tics in Tourette’s Disorder are extremely common, and

treatment strategies are directed at reducing tics as a whole in individuals with tic disorders. The

Canadian Guidelines for the Evidence Based Treatment of Tic Disorders found high quality

evidence for the efficacy of pimozide, haloperidol and risperidone for the treatment of tics,

moderate quality evidence for the efficacy of clonidine and guanfacine, low quality evidence for

the efficacy of fluphenazine, metoclopramide, aripiprazole, olanzapine, ziprasidone, topiramate,

botulinum toxin injections and cannabinoids, and very low quality of evidence for quetiapine,

baclofen, and tetrabenazine 17

. With respect to behavioral treatments, there was high quality

evidence for habit reversal therapy, and low quality evidence for exposure and response

prevention 18

.

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Another category of persistent cough which should not be confused with psychogenic cough is

unexplained cough. Unexplained, rather than idiopathic, cough was suggested by ACCP to

describe cases where behavior modification and psychiatric therapy failed to resolve cough of

unidentified organic etiology 16

. Unexplained cough should only be considered after failing both

to find a cause for cough and failing behavior modifications and psychiatric therapy 16

. A careful

evaluation should be undertaken since inadequate diagnostic workup can lead to mistakenly

over-diagnosing unexplained cough 32

.

Strengths and limitations:

The strengths of this systematic review include the comprehensive search of multiple databases

without language restriction, selecting studies in duplicate and collaborating with an

interprofessional team of content experts from the ACCP that includes pediatric, pulmonary,

neurology, otolaryngology and psychiatry expertise. To our knowledge, this is the only

systematic review on the topic.

The limitations of this review mainly relate to the lack of comparative studies, the likelihood of

publication bias, heterogeneity of terms used to describe the cough and inconsistency in

diagnostic criteria and approach across the studies. In addition, the number of patients enrolled in

the studies is small.

Implications for research and practice

The lack of comparative evidence in this chronic and burdensome disorder is compelling.

Prospective patient registries are needed for conducting rigorous observational studies to help

recruit patients in multi-center randomized controlled trials. In the absence of comparative

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evidence, the ACCP expert cough panel will extrapolate indirect evidence and incorporate the

existing evidence base with patients’ values, preferences, clinical context and availability of

therapies to guide patient care. The uncertainty in the evidence should be conveyed to patients at

the point of decision making.

Conclusion:

Only low quality evidence exists to support a particular strategy to define, diagnose and treat

psychogenic, habit and tic cough. Patient values, preferences, and availability of potential

therapies should guide treatment choice.

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Figure 1: Study selection

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Table 1: Characteristics of included studies†

Study Intervention

Study population

Number of

patients

(%males)

Age range/

mean (year)

Number of

children

Mean/median

(range) of follow-up

period

Anbar

2002 33

Hypnotherapy

254 patients with 1- pulmonary

symptoms due to psychological issues

(e.g. habit cough), 2- discomfort due to

medication or 3- fear of procedures were

agreed to be treated with hypnosis

19 (NR) 5-20 - -

Anbar

2004 20

Self-hypnosis

56 children and adolescents with habit

cough. 11% of the patients were

diagnosed with comorbid conversion

disorder.

56 (52%)

5-17/10.7 56 Mean 13 months

(range:2 months to 2

years)

Anbar

2005 34

Self-hypnosis

72 pediatric patients referred to

pulmonary center for clinical hypnosis

including those with habit cough,

anxiety, asthma, chest pain, dyspnea,

hyperventilation, sighing, and vocal cord

dysfunction

21 (NR) 10.7 21 -

Berman

1966 35

Art of suggestion

6 patients

6 (50%) 9-13 6 Reported to be 2

years in 3 patients

Cohlan

1984 21

Reinforced

suggestion

technique: bed

sheet.

33 patients 33 (39%) 5-14/9.6 33 Range 10 months to

21 years in 18

patients

Lavigne

1991 36

Suggestion

technique,

parental and self-

monitoring, and

4 children

4 (50%) 8-12/ 10 4 Range 2 weeks - 6

months

Page 17 of 41


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social or material

rewards

Lokshin

1991 37

Suggestion

therapy

9 patients, initially misdiagnosed as

asthma, were treated with brief sessions

of suggestion therapy

9 (33%) 6-17/ median

11

9 1 week to 9.4 year

(median 2.2 year)

Bernztein

1995 38

Medical advice

about family

management

83 pediatric patients who sought consult

for chronic cough diagnosis and

treatment

6 (NR) - 6 -

Bhatia

2002 11

Appropriate drug

(anxiolytic,

antidepressants)

and/or

psychotherapy

(explanation,

reassurance,

suggestion)

32 patients seen with cough in a tertiary

care teaching hospital. They were

screened by the otorhinolaryngologist

and then referred to the psychiatry

outpatient department. 62% had a

psychiatric disorder (conversion disorder

and mixed anxiety and depressive

disorder were the most common)

32 (41%) 24.2 in males

and 21.4 in

females

- -

Bordoy

2004 24

Relaxation

techniques and

psychopedagogic

support

6 patient with a diagnosis of hard-to-

manage asthma and/or persistent cough.

6 patients had comorbid generalized

anxiety disorder

6 (67%) 7-12/10.5 6 8 to 15 months

Kravitz

1969 39

Counseling,

reassurance and

tranquilizers

9 children and adolescents who missed

weeks, even months of school because of

psychogenic cough tics

9 (56%) 11-15/13 9 -

Mastrovic

h 2002 40

Decongestants,

antihistamine,

corticosteroids

and other

medications,

explaining the

nature of cough,

2 patients were referred for an evaluation

of refractory chronic cough

2 (0%) 66-89/77.5 0 7 month in one

patient, and 1 week in

the other.

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and psychiatric

referral

(paroxetine was

advised for one

patient). Throat

lozenges in one

patient

Shuper

1983 41

Psychology

counseling

6 patients 6 (67%) 6-11/9 6 Reported to be 6

month in one patient

Weinberg

1980 42

Reassurance,

tranquilizer and

referral to

psychologist

3 patients 3 (67%) 6-10/8.3 3 -

Blager

1988/Gay

1987* 23,27

Voice therapy

techniques and

psychotherapy

4 patients were admitted with a diagnosis

of asthma. 3 of the patients had comorbid

conversion disorder and the fourth

patient had somatization disorder

4 (0%) 19-30/

26.75

0 Mean 8 months,

range 6 - 12 month

after discharge

Kastelik

2005 43

Cognitive

psychotherapy

131 patients were referred for chronic

(>8 weeks) cough, two patients were

found to have psychogenic cough

2 (NR) - - -

Sipp 2007 25

Vocal fold

injection with

botulinum toxin

type A (BTX-A)

3 children with debilitating cough that

had resulted in prolonged school

absence.

3 (33%) 10-13/11.7 3 Range 9-10 month

Tan 2004 22

Haloperidol in

one patient and

no treatment in

the other

2 boys (twins) with chronic persistent

cough, one was misdiagnosed with

asthma and found to have involuntary

movements (motor tics)

2 (100%) 8 2 One year in one

patient

†All included studies are retrospective and uncontrolled

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*Both Blager 1988 and Gay 1987 reported treating the same four patients. Therefore, we included only Blager and extracted data from both

Page 20 of 41


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Table 2 Cough characteristics

Study ID Main terms used

(other terms used

interchangeably)

Description of

cough

Did cough

happen during

sleep?

Duration of

cough (mean,

range)

Triggers/

precipitating

factors

Failure of

conventional

treatment

Impact on

work/school

Anbar 2002 33

Habit cough - - - - - -

Anbar 2004 20

Habit cough (also

known as

psychogenic cough

or cough tic)

Loud (16 patients),

barky (11), croupy

(8), harsh (7), brassy

(4), disruptive (4),

honking (3), not

recorded (6)

5 patients had

cough during

sleep

13 months,

range 2 week

to 7 years

- Upper

respiratory

infections

(including otitis

media and

sinusitis) (59%)

- Asthma (13%)

- Exercise (5%)

No therapy was

helpful for the

cough in 55 patients.

One had some

improvement with

lorazepam

50% of the

patients missed at

least 1 week of

school because of

their cough

Anbar 2005 34

Habit cough - - - - - -

Berman

1966 35

Habit cough (often

referred to by

some as

psychogenic cough

or respiratory tic)

Croupy, harsh, barky

None 2-6 Months

Respiratory

infection (2

patients)

Antibiotics and anti-

tussive preparations

and other

medications were

tried with no

response

All children lost

excessive school

time

Cohlan

1984 21

Psychogenic cough

tic

Recurrent paroxysms

of barking, brassy,

foghorn, honking

cough every few

minutes

None 6.2 weeks,

range 2-24

weeks

- Cough mixtures,

antihistamines,

antibiotics and other

medications were

tried

Most of the

children denied

school attendance

because of

the continuous

disturbing noise

in the classroom

Lavigne

1991 36

Psychogenic cough -

One had

occasional

6 weeks, range

9 days - 3

Cough began

following an

Various medications

used including

2 patients had

missed school for

Page 21 of 41


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cough during

night

months

exposure to a

smoky room in

one patient and

following an a

upper respiratory

infection in

another one

antihistamines,

prednisone and

others

a few days

Lokshin

1991 37

Habit cough (We

preferred the term

"habit" cough

rather than

"psychosomatic"

or "psychogenic"

cough)

- None Median 2

month, range 1

week - 12

month

- Trials of

bronchodilators and

corticosteroids with

little or no response

-

Bernztein

1995 38

Psychogenic cough - None - - - -

Bhatia 2002 11

Psychogenic (or

nervous cough)

- - Up to 2 weeks

(4 patients), 2-

4 weeks (7

patients), 1-3

months (17

patients), more

than 3 months

(4 patients)

- School phobia

(18.8%)

- Dissatisfaction in

family (21.8%)

- Fear of rejection

and need for

attention (31.3%)

- -

Bordoy

2004 24

Psychogenic cough Dry daily

cumbersome cough

None 24.8 months - All patients failed

multiple drugs

(antibiotics,

antihistamines and

others medications)

-

Kravitz

1969 39

Psychogenic cough

tic

Explosive foghorn

type bark-like

None - Upper respiratory

tract infection (2

patients)

Cough medication

and/or antibiotics

All patients had

missed 3 weeks or

more of school

Page 22 of 41


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(range of 3-15

weeks)

Mastrovich

2002 40

Psychogenic

(habit) cough

Bouts of explosive

nonproductive cough

in one patient, and a

daily, continuous

sensation in throat

that produced

nonproductive cough

in the other patient

None Range 5 - 13

years

Cough started in

one patient after

death of her

husband, and after

having a

community-

acquired

pneumonia in the

second case

Decongestants,

antihistamines, nasal

corticosteroids and

ipratropium,

intranasal

beclomethasone

dipropionate,

prednisone, and

other medications

Normal social

activities were

disrupted in both

patients

Shuper

1983 41

Psychogenic cough Paroxysmal, harsh,

barky, non-

productive, croupy

barking bizarre

cough

None 6 month in one

patient

- Antitussive drugs 5 had lost many

school days

Weinberg

1980 42

Psychogenic cough

tic

Croupy, explosive

barking and honking

None - Upper respiratory

tract infection (3

patients)

Antibiotics and

cough suppressants

One patient had

lost many school

days

Blager

1988/Gay

1987 23,27

Habit cough

(psychogenic)

Nonproductive

hacking or barking

cough

One patient

coughed during

sleep

6.1 years,

range half

year- 16 years

Emotional stress

(3 patients)

Steroids Jobs of 2 patients

were jeopardized

because of

continuous

hospitalization

Kastelik

2005 43

Psychogenic cough - - >8 weeks - Trials of treatment

for asthma, gastro-

oesophageal reflux

and rhino-sinusitis

-

Sipp 2007 25

Habit, or

psychogenic,

cough

Nonproductive

cough with a

honking or barking

None 4 months,

range 3-6

month

Irritation in the

throat in three

patients

Antibiotics, H2-

antihistamines and

asthma inhalers

All patients

missed between 2

and

Page 23 of 41


23

quality were tried in all 3

patients without

success

6 months of

school because of

their incessant

coughing

Tan 2004 22

Chronic persistent

cough. Phonic tic

in one patient.

Nonproductive with

continuous throat

clearing

- 2 years in one

patient and 4

month in the

other one

- Antibiotics and

budesonide in one

patient

Social discomfort

was reported

Page 24 of 41


24

Table 3: Methodological quality indicators

Study ID Selection of patients

(all consecutive patients

OR some of the patients)

Percent of lost to

follow-up

Ascertainment of outcome

(records, clinic visit, phone call)

Mean/median (range) of follow-up

period

Anbar 2002 33

Not clear 5% (1 patient) By answering open-ended

questions in follow-up clinic visits

Not reported

Anbar 2004 20

A chart review identified all

patients with habit cough

4% (2 patients) Records 13 months (range: 2 months to 2

years)

Anbar 2005 34

Not clear

14.3% (3 patients ) Clinic visit Not reported

Berman 1966 35

Not clear None Not clear Reported to be 2 years in 3 patients

Cohlan 1984 21

Not clear None

Not clear Range 10 months to 21 years in 18

patients

Lavigne 1991 36

Not clear None Office visits and telephone call Range 2 weeks - 6 months

Lokshin 1991 37

Medical records of the period 1975

– 1988 were reviewed. An attempt

to contact all patients with habit

cough was made

22.2% (2 patients) Telephone interview 1 week to 9.4 year (median 2.2 year)

Bernztein 1995 38

Not clear

Not clear Clinic visit Not reported

Bhatia 2002 11

All consecutives patients over a

period of four years (1997-2001).

Patients having unreliable history

were excluded.

None Clinic visit Not reported

Page 25 of 41


25

Bordoy 2004 24

Referred patients None Not clear 8 to 15 months

Kravitz 1969 39

Not clear Not clear Not clear Not reported

Mastrovich

2002 40

Not clear Not clear Phone call in one patient and

contacting the patient’s son in the

other one

7 month in one patient, and 1 week

in the other one

Shuper 1983 41

Not clear Not clear Not clear Reported to be 6 month in one

patient

Weinberg 1980 42

Not clear

None Not clear

Not reported

Blager

1988/Gay 1987 23,27

Not clear None Contacting the patient and their

physicians after discharge

Mean 8 months, range 6 - 12 month

after discharge

Kastelik 2005 43

Retrospective evaluation of

consecutive referrals

Not clear Not clear Not clear

Sipp 2007 25

Not clear (charts were reviewed

2004 – 2006, 3 were selected)

None Records and visits Range 9-10 month

Tan 2004 22

Not clear

None Not clear

One year in one patient

Page 26 of 41


26

Table 4: Non pharmacological interventions

Hypnotherapy 3 studies (43% of patients)

Suggestion therapy with/without a distractor 4 studies (23%)

Mix of multiple interventions including reassurance, counseling,

referral to psychologist and appropriate medications

7 studies (29%)

Voice therapy techniques and psychotherapy 1 study (2%)

Cognitive psychotherapy 1 study (1%)

Vocal fold injection with botulinum toxin type A 1 study (1%)

Page 27 of 41


27

Table 5 Effects of interventions

Study ID Intervention

Pooling patients

who underwent

same intervention

Number of

patients

Results Percent of

improvement

Anbar 2002 33

Hypnotherapy

96 underwent

treatment with

hypnotherapy.

Cough resolved in

75 (78%) and

improved in 5 (5%).

19 mostly

pediatric

patients

Hypnotherapy offered to 23 patients, 19 of them

accepted, 16 improved, 2 did not improve, and 1 lost

to follow-up

84

Anbar 2004 20

Self-hypnosis

56 pediatric

patients

Among 51 patients who used hypnosis, 46 had their

cough resolved. 40 patients had cough resolved during

or immediately after the initial hypnosis instruction

session, 4 patients within 1 week, and 2 within 1

month

90

Anbar 2005 34

Self-hypnosis

21 pediatric

patients

Symptom resolved in 13 patients, and improved in 5.

The 3 other patients were lost to follow-up

86

Berman 1966 35

Art of suggestion

52 underwent

suggestion therapy

with/without a

distractor. Cough

resolved in 50

patients (96%)

6 pediatric

patients

All six children improved and have been free of cough

during a long-term period of observation

100

Cohlan 1984 21

Reinforced

suggestion

technique: bed sheet.

33 pediatric

patients

31 were successfully treated 94

Lavigne 1991 36

Suggestion

technique, parental

and self-monitoring,

and social or

material rewards

4 pediatric

patients

All 4 patients improved and the treatment resulted in

eventual elimination of the cough

100

Lokshin 1991 37

Suggestion therapy

9 pediatric

patients

All patients became symptoms free during a 15-

minute session of suggestion therapy. During the

100

Page 28 of 41


28

subsequent week, one remained completely

asymptomatic and 8 had transient minor relapses that

were readily self-controlled. At contacting the patients

for late follow-up, 6 of 7 were completely

asymptomatic, one continued to have minor self-

controlled symptoms

Bernztein

1995 38

Medical advice

about family

management

64 patients were

treated with multiple

interventions

including

appropriate

medications,

reassurance,

counseling and

referral to

psychologist.

59 (92%) patients

had their cough

stopped or

improved.

6 pediatric

patients

Symptoms improved in all 6 patients 100

Bhatia 2002 11

Appropriate drug

(anxiolytic,

antidepressants)

and/or

psychotherapy

(reassurance,

suggestion)

32 patients 12 cases remitted, 16 cases improved (decrease in

frequency of cough) and 4 cases continued coughing

88

Bordoy 2004 24

Relaxation

techniques and

psychopedagogic

support

6 pediatric

patients

In all 5 patients who received the treatment, the cough

resolved within 2-4 weeks after the intervention. In the

other patient that didn’t received the intervention

treatment, the cough disappeared after 4 weeks

without any relapse at 8 and 15 months of follow-up

100

Kravitz 1969 39

Counseling,

reassurance and

tranquilizers

9 pediatric

patients

All 9 patients responded to tranquilizers, suggestion,

reassurance and counseling

100

Mastrovich

2002 40

Decongestants,

antihistamine,

corticosteroids and

other medications,

explaining the

nature of cough, and

psychiatric referral (

2 adult

patients

The cough didn’t change in the first patient; however,

the patient was able to suppress it with a throat

lozenge. The cough improved in the second patient

although she discontinued paroxetine after 3 days.

Both patients declined follow-up service and refused

the psychiatric referral

50

Page 29 of 41


29

paroxetine was

advised for one

patient)

Shuper 1983 41

Psychology

counseling

6 pediatric

patients

Cough disappeared in all patients after few weeks of

treatment

100

Weinberg

1980 42

Reassurance,

tranquilizer and

referral to

psychologist

3 pediatric

patients

Cough improved in all 3 patients 100

Blager

1988/Gay

1987 23,27

Voice therapy

techniques and

psychotherapy

4 mostly

adult

patients

3 patients reported significant improvement with their

symptom and no emergency room visits or

hospitalizations in the follow-up

75

Kastelik

2005 43

Cognitive

psychotherapy

2 patients Cough was resolved in the two patients 100

Sipp 2007 25

Vocal fold injection

with botulinum

toxin type A (BTX-

A)

3 pediatric

patients

All 3 patients had cessation of their cough after the

injections, and all 3 reported decreased irritation in

their throats at post-treatment visits. Their coughs

recurred but were controlled with 4 to 8 sessions of

behavioral therapy

100

Tan 2004 22

Haloperidol in one

patient and no

treatment in the

other

2 pediatric

patients

Cough and other phonic tics totally disappeared in the

first twin after two weeks of haloperidol. The

symptoms were completely resolved without any

treatment in the second twin

100

Page 30 of 41


31

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166x175mm (96 x 96 DPI)

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Appendix

Ovid

Database(s): Embase 1988 to 2013 Week 36, Ovid MEDLINE(R) In-Process & Other Non-Indexed Citations and Ovid MEDLINE(R) 1946 to Present, PsycINFO 1806 to September Week 1 2013, EBM Reviews - Cochrane Central Register of Controlled Trials August 2013, EBM Reviews - Cochrane Database of Systematic Reviews 2005 to July 2013 Search Strategy:

# Searches Results

1 exp Cough/ 71072

2 cough*.tw,kw,hw,sh. 116370

3 exp Habits/ 185907

4 exp Tics/ 9594

5 exp psychosomatic disorder/ 52121

6 exp Psychophysiologic Disorders/ 52121

7 (tic or tics or habit* or psychogenic* or "psycho organic*" or psychoautonomic* or psychoorganic* or psychophysiologic* or psychosomatic* or psychosomatos* or somatopsychic*).tw,kw,hw,sh.

417464

8 or/3-7 577828

9 (1 or 2) and 8 3298

10 cough/px 149

11 ("vocal tic" or "vocal tics").mp. [mp=ti, ab, sh, hw, tn, ot, dm, mf, dv, kw, nm, kf, ps, rs, ui, tc, id, tm, tx, ct]

1277

12 9 or 10 or 11 4637

13 exp Pimozide/ 6422

14 exp haloperidol/ 55624

15 exp Fluphenazine/ 8016

16 exp metoclopramide/ 21222

17 exp Risperidone/ 34718

18 exp aripiprazole/ 9061

19 exp Antipsychotic Agents/ 272836

20 exp olanzapine/ 26333

21 exp quetiapine/ 16235

22 exp ziprasidone/ 6651

23 exp clonidine/ 40735

24 exp Guanfacine/ 2407

25 exp topiramate/ 14072

26 exp Baclofen/ 17437

27 exp botulinum toxin/ 23248

28 exp Tetrabenazine/ 2612

29 exp Cannabinoids/ 47252

30 exp Suggestion/ 4458

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31 exp Psychotherapy/ 465399

32 exp Counseling/ 188199

33 exp Mind-Body Therapies/ 76366

34 exp Speech Therapy/ 16379

35 exp self monitoring/ 5911

36 exp Deep Brain Stimulation/ 25571

37 exp Transcranial Magnetic Stimulation/ 22680

38 exp Neuroleptic Drugs/ 153216

39 exp biofeedback/ 71036

40 exp behavior modification/ 104077

41

(therap* or treatment* or intervention* or antipsychotic* or "anti-psychotic*" or neuroleptic* or Pimozide or orap or antalon or Halloperidol or haloperidol or haldol or fluphenazine or lyogen or prolixin or flufenazin or metoclopramide or primperan or maxolon or methoxybenzamide or cerucal or rimetin or reglan or Risperidone or risperdal or Aripiprazole or Olanzapine or Quetiapine or Ziprasidone or Clonidine or clofelin or klofelin or catapres* or clopheline or klofenil or isoglaucon or clofenil or hemiton or chlophazolin or gemition or dixarit or Guanfacine or tenex or topiramate or Baclofen* or genbaclofen* or apobaclofen* or baclophen* or Botulin* or botox or Tetrabenazine or xenazine or nitoman or Cannabinoid* or suggestion* or "Aversion therap*" or "aversive therap*" or (behavior* adj2 modif*) or counseling or "bed sheet*" or hypnosis or hypnot* or hypnotherap* or biofeedback* or hypnoses or mesmerism or "autogenic training" or feedback* or myofeedback* or neurofeedback* or "speech therap*" or (habit* adj2 reversal) or "massed negative practice*" or "self-monitor*" or "Contingency management" or (exposure* adj2 response*) or (brain adj3 stimulation*) or "Transcranial Magnetic Stimulation*").mp. [mp=ti, ab, sh, hw, tn, ot, dm, mf, dv, kw, nm, kf, ps, rs, ui, tc, id, tm, tx, ct]

12315772

42 exp Cough/dh, dt, pc, rt, su, th [Diet Therapy, Drug Therapy, Prevention & Control, Radiotherapy, Surgery, Therapy]

2974

43 exp coughing/dm, dt, rt, su, th [Disease Management, Drug Therapy, Radiotherapy, Surgery, Therapy]

7824

44 or/13-43 12510011

45 12 and 44 2008

46 exp controlled study/ 4222876

47 exp evidence based medicine/ 685107

48 evidence-based.mp. 255646

49 ((control$ or randomized) adj2 (study or studies or trial or trials)).mp. [mp=ti, ab, sh, hw, tn, ot, dm, mf, dv, kw, nm, kf, ps, rs, ui, tc, id, tm, tx, ct]

5520709

50 meta analysis/ 129236

51 meta-analys$.mp. 219593

52 exp "systematic review"/ 63835

53 (systematic* adj review$).mp. 166233

54 exp Guideline/ or exp Practice Guideline/ 328683

55 guideline$.ti. 111337

56 exp case study/ 1691935

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57 follow up studies/ 1229677

58 exp Cohort Studies/ 1607717

59 exp longitudinal study/ 1028472

60 exp retrospective study/ 818121

61 exp prospective study/ 671928

62 exp observational study/ 44013

63 exp comparative study/ 2494726

64 exp clinical trial/ 1736823

65 exp evaluation/ 1352063

66 exp twins/ 51652

67 exp validation study/ 42414

68 exp experimental study/ or exp field study/ or in vivo study/ or exp panel study/ or exp pilot study/ or exp prevention study/ or exp quasi experimental study/ or exp replication study/ or exp theoretical study/ or exp trend study/ or clinical study/

1767624

69

((clinical or evaluation or twin or validation or experimental or field or "in vivo" or panel or pilot or prevention or replication or theoretical or trend or comparative or cohort or longitudinal or retrospective or prospective or population or concurrent or incidence or follow-up or observational or multivariate) adj (study or studies or survey or surveys or analysis or analyses or trial or trials)).mp.

8417404

70 ("case study" or "case series" or "clinical series" or "case studies").mp. [mp=ti, ab, sh, hw, tn, ot, dm, mf, dv, kw, nm, kf, ps, rs, ui, tc, id, tm, tx, ct]

271953

71 exp Case-Control Studies/ 752470

72 "case control*".mp. 348642

73 study.tw. 11305277

74 or/46-73 20814225

75 45 and 74 1292

76 from 45 keep 892-1914 1023

77

limit 76 to (clinical trial, all or clinical trial, phase i or clinical trial, phase ii or clinical trial, phase iii or clinical trial, phase iv or clinical trial or comparative study or controlled clinical trial or evaluation studies or guideline or meta analysis or multicenter study or practice guideline or randomized controlled trial or systematic reviews or twin study or validation studies) [Limit not valid in Embase,PsycINFO,CCTR,CDSR; records were retained]

177

78 75 or 77 1293

79

limit 78 to (book or book series or editorial or erratum or letter or note or addresses or autobiography or bibliography or biography or comment or dictionary or directory or interactive tutorial or interview or lectures or legal cases or legislation or news or newspaper article or overall or patient education handout or periodical index or portraits or published erratum or video-audio media or webcasts) [Limit not valid in Embase,Ovid MEDLINE(R),Ovid MEDLINE(R) In-Process,PsycINFO,CCTR,CDSR; records were retained]

62

80 78 not 79 1231

81 from 45 keep 1915-2008 94

82 80 or 81 1275

83 animals/ not humans/ 4646363

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84 82 not 83 1273

85 remove duplicates from 84 905

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Scopus

1 TITLE-ABS-KEY( (tic or tics or habit* or psychogenic* or "psycho organic*" or

psychoautonomic* or psychoorganic* or psychophysiologic* or psychosomatic* or

psychosomatos* or somatopsychic*) and cough*)

2 TITLE-ABS-KEY( "vocal tic" or "vocal tics")

3 1 or 2

4 TITLE-ABS-KEY(therap* or treatment* or intervention* or antipsychotic* or "anti-

psychotic*" or neuroleptic* or Pimozide or orap or antalon or Halloperidol or haloperidol or

haldol or fluphenazine or lyogen or prolixin or flufenazin or metoclopramide or primperan or

maxolon or methoxybenzamide or cerucal or rimetin or reglan or Risperidone or risperdal or

Aripiprazole or Olanzapine or Quetiapine or Ziprasidone or Clonidine or clofelin or klofelin

or catapres* or clopheline or klofenil or isoglaucon or clofenil or hemiton or chlophazolin or

gemition or dixarit or Guanfacine or tenex or topiramate or Baclofen* or genbaclofen* or

apobaclofen* or baclophen* or Botulin* or botox or Tetrabenazine or xenazine or nitoman or

Cannabinoid* or suggestion* or "Aversion therap*" or "aversive therap*" or (behavior* W/2

modif*) or counseling or "bed sheet*" or hypnosis or hypnot* or hypnotherap* or

biofeedback* or hypnoses or mesmerism or "autogenic training" or feedback* or

myofeedback* or neurofeedback* or "speech therap*" or (habit* W/2 reversal) or "massed

negative practice*" or "self-monitor*" or "Contingency management" or (exposure* W/2

response*) or (brain W/3 stimulation*) or "Transcranial Magnetic Stimulation*")

5 TITLE-ABS-KEY( (evidence W/1 based) OR (meta W/1 analys*) OR (systematic* W/2

review*) OR guideline OR (control* W/2 stud*) OR (control* W/2 trial*) OR (randomized

W/2 stud*) OR (randomized W/2 trial*) or "comparative study" OR "comparative survey"

OR "comparative analysis" OR "cohort study" OR "cohort survey" OR "cohort analysis" OR

"longitudinal study" OR "longitudinal survey" OR "longitudinal analysis" OR "retrospective

study" OR "retrospective survey" or "retrospective analysis" OR "prospective study" OR

"prospective survey" OR "prospective analysis" OR "population study" OR "population

survey" OR "population analysis" OR "concurrent study" OR "concurrent survey" OR

"concurrent analysis" or "incidence study" OR "incidence survey" OR "incidence analysis"

OR "follow-up study" OR "follow-up survey" OR "follow-up analysis" or "observational

study" OR "observational survey" OR "observational analysis" OR "case study" OR "case

series" OR "clinical series" OR "case studies" or "clinical study" OR "clinical trial" or

"evaluation study" OR "evaluation survey" OR "evaluation analysis" or "twin study" OR

"twin survey" OR "twin analysis" or "validation study" OR "validation survey" OR

"validation analysis" or "experimental study" OR "experimental analysis" or "field study" OR

"field survey" OR "field analysis" or "in vivo study" OR "in vivo analysis" or "panel study"

OR "panel survey" OR "panel analysis" or "pilot study" OR "pilot survey" OR "pilot

analysis" or "prevention study" OR "prevention survey" OR "prevention analysis" or

"replication study" OR "replication analysis" or "theoretical study" OR "theoretical analysis"

or "trend study" OR "trend survey" OR "trend analysis" or "case control*" or "multivariate

stud*" or "multivariate analys*" or study)

6 3 and 4 and 5

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7 PMID(0*) OR PMID(1*) OR PMID(2*) OR PMID(3*) OR PMID(4*) OR PMID(5*) OR

PMID(6*) OR PMID(7*) OR PMID(8*) OR PMID(9*)

8 6 and not 7

9 DOCTYPE(le) OR DOCTYPE(ed) OR DOCTYPE(bk) OR DOCTYPE(er) OR

DOCTYPE(no) OR DOCTYPE(sh)

10 8 and not 9

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Management and Diagnosis of Psycogenic Cough, Habit Cough, and Tic Cough: A Systematic Review

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