Management and diagnosis of psychogenic cough, habit cough, and tic cough: a systematic review.

[

Original Research Signs and Symptoms of Chest Diseases

]

Management and Diagnosis of Psychogenic Cough, Habit Cough, and Tic Cough A Systematic Review Qusay Haydour, MD; Fares Alahdab, MD; Magdoleen Farah, MBBS; Patricia Barrionuevo, MD; Anne E. Vertigan, PhD; Peter A. Newcombe, PhD; Tamara Pringsheim, MD; Anne B. Chang, PhD; Bruce K. Rubin, MD; Lorcan McGarvey, MD; Kelly A. Weir, BSpThy, MSpPath; Kenneth W. Altman, MD, PhD; Anthony Feinstein, PhD; Mohammad Hassan Murad, MD, MPH; and Richard S. Irwin, MD, Master FCCP

Several pharmacologic and nonpharmacologic therapeutic options have been used to treat cough that is not associated with a pulmonary or extrapulmonary etiology.

BACKGROUND:

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.

A total of 18 uncontrolled studies were identified, enrolling 223 patients (46% male subjects, 96% children and adolescents). Psychogenic cough was the most common descriptive term used (90% of the studies). Most of the patients (95%) had no cough during sleep; barking or honking quality of cough was described in only eight studies. Hypnosis (three studies), suggestion therapy (four studies), and counseling and reassurance (seven 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. RESULTS:

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 CHEST 2014; 146(2):355-372 therapies should guide treatment choice.

CONCLUSIONS:

Manuscript received April 1, 2014; revision accepted April 21, 2014; originally published Online First May 15, 2014. ABBREVIATIONS: CHEST 5 American College of Chest Physicians; DSM 5 Diagnostic and Statistical Manual of Mental Disorders AFFILIATIONS: From the Mayo Clinic (Drs Haydour, Alahdab, Farah, Barrionuevo, and Murad), The Knowledge and Evaluation Research Unit and the Center for the Science of Health Care Delivery, Rochester, MN; Unidad de Conocimiento y Evidencia (Dr Barrionuevo), Universidad Peruana Cayetano Heredia, Lima, Peru; John Hunter Hospital (Dr Vertigan), Department of Speech Pathology, Newcastle, NSW, Australia; University of Queensland (Dr Newcombe), School of Psychology, Brisbane, QLD, Australia; University of Calgary (Dr Pringsheim),

journal.publications.chestnet.org

Calgary, AB, Canada; Royal Children’s Hospital and Menzies School of Health Research (Dr Chang), Charles Darwin University, Brisbane, Brisbane, QLD, Australia; Children’s Hospital of Richmond and Virginia Commonwealth University (Dr Rubin), Richmond, VA; Centre for Infection and Immunity (Dr McGarvey), The Queen’s University of Belfast, Belfast, Northern Ireland; Royal Children’s Hospital Department of Speech Pathology and Queensland Children’s Medical Research Institute (Ms Weir), The University of Queensland, Brisbane, QLD, Australia; Mount Sinai Hospital (Dr Altman), New York, NY; Sunnybrook Health Sciences Centre (Dr Feinstein), Toronto, ON, Canada; and UMass Memorial Medical Center (Dr Irwin), Worcester, MA.

355

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 and 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 psychologic and neurologic 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 (eg, gastroesophageal reflux disease) etiology in the presence of some suggestive clinical characteristics and/or an association with psychologic issue. However, these terms

Materials and Methods This systematic review follows an a priori established protocol developed by the guideline methodologist and selected members of the CHEST 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 or more patients 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,

The authors have reported to CHEST that no funding was received for this study. CORRESPONDENCE TO: Qusay Haydour, MD, 257 Napa Dr, Augusta, GA 30909; e-mail: [email protected] © 2014 AMERICAN COLLEGE OF CHEST PHYSICIANS. Reproduction of this article is prohibited without written permission from the American College of Chest Physicians. See online for more details. DOI: 10.1378/chest.14-0795 FUNDING/SUPPORT:

356 Original Research

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 nighttime.9-12 Holinger and Sanders13 found that psychogenic cough was the second most common cause of chronic cough in children aged 6 to 16 years. Many interventions, including pharmacotherapy, behavioral modifications, and psychotherapy, were used, but none has 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 postviral vagal neuropathy (which is considered more strictly to be a disorder of the peripheral receptors or brainstem reflex feedback loop). The American College of Chest Physicians (CHEST) develops clinical practice guidelines to help patients and physicians in decision-making. To aid in 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.

Ovid MEDLINE, Ovid EMBASE, Ovid PsycInfo, Ovid Cochrane Central 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 CHEST expert panel. The search used both controlled vocabulary and keywords. The strategy used is described in e-Appendix 1. The reference lists from the narrative reviews and existing guidelines16-18 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 k statistic for full text screening (k 5 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 NewcastleOttawa 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 Internet-based form to extract for each eligible study the following data

[

146#2 CHEST AUGUST 2014

]

items: study design, study population, study main objective, number of patients, age, sex, 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 with or failed the treatment.

Results

term psychogenic cough tic was mentioned in three studies. None of the 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.36 None of the studies used any validated cough assessment tool. There were no clear diagnostic criteria followed in any of the studies other than describing the symptoms with which the patients presented. Nonproductive explosive barking or honking cough were the most common reported cough features, but these characteristics were only reported in eight of the studies. Five percent of patients (seven of 147 patients for whom this information was reported) had cough during sleep. Duration of cough varied widely among patients, with a range of 2 weeks to 16 years. About one-half of the studies reported triggers and precipitating factors, of which upper respiratory infections and social distress were the most predominant (Table 2).

Characteristics of Included Studies

The study selection process is depicted in Figure 1. This evidence summary included 223 patients enrolled in 18 uncontrolled studies published between 1966 and 2007. Of the 223 patients, 81 were male (46% of patients where sex was reported). Of the 18 studies, 13 enrolled children and adolescents only and two 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 one-half of the 223 patients were reported in three larger case series: Anbar and Hall,20 Bhatia et al,11 and Cohlan and Stone.21 The characteristics of included studies are summarized in Table 1. The description of cough is available in Table 2. The methodologic quality indicators of the studies are in Table 3. In general, the studies were uncontrolled and considered to be at high risk for bias. Cough Characteristics

Psychogenic cough was the term used in . 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

Four studies11,20,28,31 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 et al11 reported that 20 patients (62% of total patients) had comorbid psychiatric disorders that were diagnosed according to International Classification of Diseases, 10th revision; the three most common were conversion disorder (22%), mixed anxiety and depressive disorder (12%), and generalized anxiety disorder (10%). Anbar and Hall20 reported that conversion disorder was diagnosed in six patients (11% of total patients) and anxiety disorder in one patient (2%). Blager et al31 reported that three patients (75%) in their study met the criteria for the Diagnostic and Statistical Manual of Mental Disorders (DSM)-3 of conversion disorder and one patient met the criteria for somatization disorder. Bordoy et al28 reported that all six patients in their study had generalized anxiety disorder. The social and personal burdens of this cough on children were reported in eight studies in which . 69 patients missed a period of school attendance ranging from a few days to 6 months. The professional and social lives of four adult patients were also affected and disturbed. Interventions

Figure 1 – Study selection.


Fourteen studies reported using conventional pharmacologic interventions (mostly in children) prior to the 357


[


]

Hypnotherapy

Self-hypnosis

Self-hypnosis

Art of suggestion

Reinforced suggestion technique: bed sheet

Suggestion technique, parental and self-monitoring, and social or material rewards

Suggestion therapy

Medical advice about family management

Anbar and Hall20

Anbar and Hummell23

Berman24

Cohlan and Stone21

Lavigne et al25

Lokshin et al26

Bernztein and Grenoville27

Intervention

Study Population

83 pediatric patients who sought consultation for chronic cough diagnosis and treatment

6 (NR)

9 (33)

4 (50)

4 children

9 patients, initially misdiagnosed as asthma, were treated with brief sessions of suggestion therapy

33 (39)

…

6-17 (median, 11)

8-12 (10)

5-14 (9.6)

9-13

10.7

21 (NR)

6 (50)

5-17 (10.7)

5-20

Age Range (mean), y

56 (52)

19 (NR)

Patients, No. (% Male)

33 patients

6 patients

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

56 children and adolescents with habit cough; 11% were diagnosed with comorbid conversion disorder.

254 patients agreed to be treated with hypnosis for (1) pulmonary symptoms due to psychologic issues (eg, habit cough), (2) discomfort due to medication, or (3) fear of procedures

] Characteristics of Included Studiesa

Anbar22

Study

TABLE 1

6

9

4

33

6

21

56

…

Children, No.

…

(Continued)

Median 2.2 y (1 wk to 9.4 y)

(2 wk to 6 mo)

(10 mo to 21 yr in 18 patients)

Reported to be 2 yr in 3 patients

…

Mean 13 mo (2 mo to 2 y)

…

Follow-up Period, Mean/Median (Range)


359

Relaxation techniques and psychopedagogic support

Counseling, reassurance, and tranquilizers

Decongestants, antihistamines, corticosteroids, and other medications, explaining the nature of cough, and psychiatric referral (paroxetine was advised for 1 patient). Throat lozenges in 1 patient

Psychology counseling

Reassurance, tranquilizer, and referral to psychologist

Bordoy et al28

Kravitz et al29

Mastrovich and Greenberger10

Shuper et al30

Weinberg9

Intervention

Appropriate drug (anxiolytic, antidepressants) and/or psychotherapy (explanation, reassurance, suggestion)

] (continued)

Bhatia et al11

Study

TABLE 1

6 (67) 3 (67)

3 patients

6-10 (8.3)

6-11 (9)

66-89 (77.5)

2 (0)

2 patients were referred for an evaluation of refractory chronic cough

6 patients

11-15 (13)

9 (56)

9 children and adolescents who missed weeks, even months, of school because of psychogenic cough tics

Male patients: 24.2; female patients: 21.4

Age Range (mean), y

7-12 (10.5)

32 (41)


6 (67)

6 patients with a diagnosis of hard-to-manage asthma and/or persistent cough. 6 patients had comorbid generalized anxiety disorder.

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).

Study Population

3

6

0

9

…

…

(Continued)

Reported to be 6 mo in 1 patient

7 mo in one patient, 1 wk in the other

(8-15 mo)

…

…

6


Children, No.


[


]

Vocal fold injection with botulinum toxin type A

Haloperidol in 1 patient and no treatment in the other

Sipp et al34

Tan et al35 2 boys (twins) with chronic persistent cough; 1 was misdiagnosed with asthma and found to have involuntary movements (motor tics)

3 children with debilitating cough that had resulted in prolonged school absence

131 patients were referred for chronic (. 8 wk) cough, 2 were found to have psychogenic cough

4 patients were admitted with a diagnosis of asthma. 3 had comorbid conversion disorder and the fourth patient had somatization disorder.

Study Population

10-13 (11.7)

8

2 (100)

…

19-30 (26.75)

Age Range (mean), y

3 (33)

2 (NR)

4 (0)


NR 5 not reported. All included studies were retrospective and uncontrolled. bBoth Blager et al31 and Gay et al32 reported treating the same four patients. Therefore, we included only Blager et al31 and extracted data from both.

a

Cognitive psychotherapy

Kastelik et al33

Intervention

Voice therapy techniques and psychotherapy

] (continued)

Blager et al31; Gay et al32, b

Study

TABLE 1

2

3

…

0

Children, No.

1 y in 1 patient

(9-10 mo)

…

Mean 8 mo (6-12 mo after discharge)


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 pharmacologic interventions and focused on nonpharmacologic interventions. In terms of antipsychotic medications, Tan et al35 reported two boys (twins) with chronic cough that turned out to be phonic tic of Tourette syndrome. One of the boys was successfully treated with haloperidol, whereas cough spontaneously resolved in the other boy. Nonpharmacologic 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 used 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 used a mix of interventions, including reassurance, counseling, relaxation techniques, referral to a psychologist, psychotherapy, and appropriate medications (tranquilizers, anxiolytic, and antidepressants). Of patients who received these interventions, 93% showed improvement in cough. Vocal-Fold Injection With Botulinum Toxin Type A

Sipp et al34 reported treating three cases of debilitating cough with vocal-fold injection of botulinum toxin type A. The treatment was reported as effective in breaking the cough cycle in all three children. However, the cough recurred after a period of time ranging from 10 days to 2½ months and behavioral therapy was successful in controlling the cough afterward.


Voice Therapy Techniques and Cognitive Psychotherapy

Blager et al31 showed that voice therapy techniques and psychotherapy were effective in improving cough in three of the four adult patients who received the intervention. Cognitive psychotherapy was reported in one study to be an effective treatment of 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 lowquality evidence supporting all the strategies used in the studies, because of the uncontrolled (noncomparative) nature of each of the studies.36 In addition, we consider the presence of reporting bias likely to be very high. In general, pharmacologic interventions were reported to be ineffective. Several nonpharmacologic strategies were reported to be effective. Suggestion therapy techniques essentially referred to the process of the physician convincing the patient that he or she can control the cough by resisting the urge to cough. The physician explains the nature of the cough to the patient and the patient’s family and expresses confidence in the patient’s ability to stop the cough. Distractors of a bed sheet wrapped around the patient’s chest or nebulized lidocaine diluted in normal saline have been used during the process. The role of the distractor is to help 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 predominate in many of the reported cases. It has been reported that a barking or honking quality of cough and absence of cough at night are suggestive of psychogenic, habit, and tic cough.32 However, barking or honking cough was only present in eight of the studies and nighttime coughing was reported in three studies in this review. A prospective study in adults showed that the absence of cough during sleep is not diagnostically helpful because other etiologies (eg, chronic bronchitis and emphysema) can present with the same feature.37 Moreover, in another prospective study in adults,

361


[


]

Habit cough

Habit cough (often referred to by some as psychogenic cough or respiratory tic)

Psychogenic cough tic

Anbar and Hummell23

Berman24

Cohlan and Stone21

Psychogenic cough

Habit cough (also known as psychogenic cough or cough tic)

Anbar and Hall20

Lavigne et al25

Habit cough

Main Terms Used (Other Terms Used Interchangeably)

] Cough Characteristics

Anbar22

Study

TABLE 2

…

Did Cough Happen During Sleep?

…

Recurrent paroxysms of barking, brassy, foghorn, honking cough every few minutes

Croupy, harsh, barky

…

One had occasional cough during night

None

None

…

5 patients Loud had cough (16 patients), during barky (11), sleep croupy (8), harsh (7), brassy (4), disruptive (4), honking (3), not recorded (6)

…

Description of Cough

6 wk (9 d to 3 mo)

6.2 wk (2-24 wk)

2-6 mo

…

13 mo (2 wk to 7 y)

…

Duration of Cough, Mean, (Range)

…

No therapy was helpful for the cough in 55 patients. One had some improvement with lorazepam.

…

Failure of Conventional Treatment

Cough began following an exposure to a smoky room in 1 patient and following an a URI in another

…

Various medications used, including antihistamines and prednisone

Cough mixtures, antihistamines, antibiotics, and other medications were tried

Respiratory infection Antibiotics, antitussive in 2 patients preparations, and other medications were tried with no response

…

URIs, including otitis media and sinusitis (59); asthma (13); exercise (5)

…

Triggers or Precipitating Factors (%)

(Continued)

Two patients had missed school for a few days

Most of the children denied school attendance because of the continuous disturbing noise in the classroom

All children lost excessive school time

…

50% of the patients missed at least 1 wk of school because of their cough

…

Impact on Work or School


363

26

…

Psychogenic (or nervous cough)

Psychogenic cough


Psychogenic (habit) cough

Bhatia et al11

Bordoy et al28

Kravitz et al29

Mastrovich and Greenberger10 Bouts of explosive nonproductive cough in 1 patient, and a daily, continuous sensation in throat that produced nonproductive cough in the other patient

Explosive foghorn type bark-like

Dry daily cumbersome cough

…

Psychogenic cough

…



Habit cough (preferred the term “habit” cough rather than “psychosomatic” or “psychogenic” cough)


] (continued)

Lokshin et al

Study

TABLE 2

(5-13 y)

None

24.8 mo

Up to 2 wk (4 patients), 2-4 wk (7 patients), 1-3 mo (17 patients), . 3 mo (4 patients)

…

…

…

Median 2 mo (1 wk to 12 mo)


None

None

None

None


Cough started in 1 patient after death of her husband, and after having a communityacquired pneumonia in the second case

URI in 2 patients

Normal social activities were disrupted in both patients Decongestants, antihistamines, nasal corticosteroids and ipratropium, intranasal beclomethasone dipropionate, prednisone, and other medications

(Continued)

All patients had missed ⱖ 3 wk of school (3-15 wk)

…

Cough medication and/or antibiotics

All patients failed multiple drugs (antibiotics, antihistamines and others medications)

…

…

School phobia (18.8); dissatisfaction in family (21.8); fear of rejection and need for attention (31.3) …

…

…


…

Trials of bronchodilators and corticosteroids, with little or no response


…

…



[


]

30

Psychogenic cough

Habit or psychogenic cough

Kastelik et al33

Sipp et al34

URI 5 upper respiratory tract infection.

Chronic persistent cough. Phonic tic in 1 patient.

Habit cough (psychogenic)

Blager et al31; Gay et al32

Tan et al35


Weinberg9

Psychogenic cough


] (continued)

Shuper et al

Study

TABLE 2


Nonproductive with continuous throat clearing

Nonproductive cough with a honking or barking quality

…

Nonproductive hacking or barking cough

Croupy, explosive, barking, and honking

None

…

…

One patient coughed during sleep

None

None Paroxysmal, harsh, barky, nonproductive, croupy, barking bizarre cough


URI in 3 patients

…


2 y in 1 patient, 4 mo in the other

4 mo (3-6 mo)

. 8 wk

…

Irritation in the throat in 3 patients

…

6.1 y Emotional stress in (6 mo to 16 y) 3 patients

…

6 mo in 1 patient


Antibiotics and budesonide in 1 patient

Antibiotics, H2antihistamines, and asthma inhalers were tried in all 3 patients without success

Trials of treatment of asthma, gastroesophageal reflux, and rhinosinusitis

Steroids

Antibiotics and cough suppressants

Antitussive drugs


Social discomfort was reported

All patients missed 2-6 mo of school because of their incessant coughing

…

Jobs of 2 patients were jeopardized because of continuous hospitalization

One patient had lost many school days

Five had lost many school days


TABLE 3

] Methodologic Quality Indicators Selection of Patients (All Consecutive Patients or Some Patients)

Study

Lost to Follow-up, % (No.)

Ascertainment of Outcome


Anbar22

Not clear

5 (1)

By answering open-ended questions in follow-up clinic visits

Not reported

Anbar and Hall20

A chart review identified all patients with habit cough

4 (2)

Records

13 mo (2 mo to 2 y)

Anbar and Hummell23

Not clear

Clinic visit

Not reported

Berman24

Not clear

None

Not clear

Reported to be 2 y in 3 patients

Cohlan and Stone21

Not clear

None

Not clear

(10 mo to 21 y in 18 patients)

Lavigne et al25

Not clear

None

Office visits and telephone call

(2 wk to 6 mo)

Lokshin et al26

Medical records of 1975-1988 were reviewed. An attempt to contact all patients with habit cough was made.

22.2 (2)

Telephone interview

Median 2.2 y (1 wk to 9.4 y)


Not clear

Not clear

Clinic visit

Not reported

Bhatia et al11

All consecutives patients over 4 y (1997-2001). Patients having unreliable history were excluded.

None

Clinic visit

Not reported

Bordoy et al28

Referred patients

None

Not clear

(8-15 mo)

Kravitz et al

Not clear

Not clear

Not clear

Not reported

Mastrovich and Greenberger10

Not clear

Not clear

Phone call for 1 patient and contacting the patient’s son in the other one

7 mo in 1 patient, 1 wk in the other

Shuper et al30

Not clear

Not clear

Not clear

Reported to be 6 mo in 1 patient

Weinberg9

Not clear

None

Not clear

Not reported


Not clear

None

Contacting the patient and their physicians after discharge

Mean 8 mo (6-12 mo after discharge)

Kastelik et al33

Retrospective evaluation of consecutive referrals

Not clear

Not clear

Sipp et al34

Not clear (charts were reviewed for years 2004-2006; 3 were selected)

None

Records and visits

(9-10 mo)

Tan et al35

Not clear

None

Not clear

1 y in 1 patient

29

14.3 (3)

Not clear

honking and barking characteristics were also found not to be useful in diagnosing the cause of cough.38 Given the lack of both specific clinical features and diagnostic tests, cases of psychogenic cough have become essenjournal.publications.chestnet.org

tially 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. 365

TABLE 4

] Nonpharmacologic Interventions

Intervention

Studies, No. (%)a

Hypnotherapy

3 (43)

Suggestion therapy with/without a distractor

4 (23)

Mix of multiple interventions, including reassurance, counseling, referral to psychologist, and appropriate medications

7 (29)


1 (2)


1 (1)


1 (1)

This represents patients’ percent of the total 223 patients.

a

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 that are incompatible with recognized neurologic 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 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 neurologic function, so if cough is viewed in this light, this would be the more correct of the two nomenclatures.39,40 Similarly, the term “habit disorders,” which previously was used to refer to tic disorders, trichotillomania, complex motor stereotypes, 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 syndrome or chronic vocal tic disorder, there is evidence supporting both pharmacologic therapy and behavioral therapy for tics. Coughing tics in Tourette syndrome 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 366 Original Research

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 Another category of persistent cough, which should not be confused with psychogenic cough, is unexplained cough. Unexplained, rather than idiopathic, cough was suggested by CHEST 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 overdiagnosing unexplained cough.41 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 CHEST 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

[


]

TABLE 5

] Effects of Interventions

Study

Intervention

Pooling Patients Who Underwent Same Intervention

Patients No. (Age Group)

Results

Improvement, %

96 underwent treatment with hypnotherapy. Cough resolved in 75 (78%) and improved in 5 (5%).

19 (mostly children)

Hypnotherapy offered to 23 patients: 19 accepted; 16 improved, 2 did not improve, and 1 was lost to follow-up

84

Anbar and Hall20 Self-hypnosis


56 (children)

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 wk, and 2 within 1 mo

90

Anbar and Hummell23

Self-hypnosis


21 (children)

Symptom resolved in 13 patients and improved in 5. The 3 other patients were lost to follow-up.

86

Berman24

Art of suggestion

52 underwent suggestion therapy with/without a distractor. Cough resolved in 50 patients (96%).

6 (children)

All 6 children improved and have been free of cough during a long-term period of observation

Cohlan and Stone21

Reinforced suggestion technique: bed sheet.


33 (children)

31 were successfully treated

Lavigne et al25

Suggestion technique, parental and self-monitoring, and social or material rewards


4 (children)

All 4 patients improved and the treatment resulted in eventual elimination of the cough

Anbar22

Hypnotherapy

100

94

100

(Continued)


367

TABLE 5

] (continued)

Study

Intervention



Results

Improvement, %

Lokshin et al26

Suggestion therapy


9 (children)

All patients became symptom free during a 15-min session of suggestion therapy. During the subsequent week, 1 remained completely asymptomatic and 8 had transient minor relapses that were readily self-controlled. At late follow-up, 6 of 7 were completely asymptomatic, 1 continued to have minor self-controlled symptoms.

100


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 (children)

Symptoms improved in all 6 patients

100

Bhatia et al11

64 patients Appropriate were treated drug with multiple (anxiolytic, interventions, antidepressants) including and/or appropriate psychotherapy medications, (reassurance, reassurance, suggestion) counseling, and referral to psychologist. 59 (92%) patients had their cough stopped or improved.

32

12 cases remitted, 16 cases improved (decrease in frequency of cough), and 4 cases continued coughing

88

(Continued)


[


]

TABLE 5

] (continued)

Study

Intervention



Results

Improvement, %

Bordoy et al28

64 patients Relaxation were treated techniques and with multiple psychopedagogic interventions, support including appropriate medications, reassurance, counseling, and referral to psychologist. 59 (92%) patients had their cough stopped or improved.

6 (children)

In all 5 patients who received the treatment, the cough resolved within 2-4 wk after the intervention. In the other patient who did not receive the intervention, the cough disappeared after 4 wk without any relapse at 8 and 15 mo of follow-up.

100

Kravitz et al29

Counseling, reassurance, and tranquilizers


9 (children)

All 9 patients responded to tranquilizers, suggestion, reassurance, and counseling

100

Mastrovich and Decongestants, Greenberger10 antihistamine, corticosteroids, and other medications; explaining the nature of cough, and psychiatric referral (paroxetine was advised for 1 patient)


2 (adults)

The cough did not 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 the patient discontinued paroxetine after 3 d. Both patients declined follow-up service and refused the psychiatric referral.

50

(Continued)


369

TABLE 5

] (continued)

Study

Intervention



Results

Improvement, %

Shuper et al30

Psychology counseling


6 (children)

Cough disappeared in all patients after few weeks of treatment

100

Weinberg9

Reassurance, tranquilizer, and referral to psychologist


3 (children)

Cough improved in all 3 patients

100



…

4 (mostly adults)

3 patients reported significant improvement with their symptom and no ED visits or hospitalizations in the follow-up

Kasteliket al33


…

2

Cough was resolved in the 2 patients

100

Sipp et al34


…

3 (children)

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 et al35

Haloperidol in 1 patient and no treatment in the other

…

2 (children)

Cough and other phonic tics totally disappeared in the first twin after 2 wk of haloperidol. The symptoms were completely resolved without any treatment in the second twin.

100


[

75


]

observational studies to help recruit patients in multicenter, randomized controlled trials. In the absence of comparative evidence, the CHEST 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

Acknowledgments Author contributions: Q. H. is the guarantor and takes full responsibility for the accuracy and integrity of the data in this manuscript. Q. H., F. A., M. F., and P. B. contributed to screening of abstracts and full texts and data extraction; Q. H. and M. H. M. contributed to drafting of the manuscript; Q. H., F. A., M. F., P. B., A. E. V., P. A. N., T. P., A. B. C., B. K. R., L. M., K. A. W., K. W. A., A. F., M. H. M., and R. S. I. contributed to critical revision of the manuscript for important intellectual content. Financial/nonfinancial disclosures: The authors have reported to CHEST the following conflicts of interest: In the last 3 years, Dr Altman was a consultant for Nestlé SA and Watermark Research Partners Inc (for Stryker Corp). He has no conflict of interest in the last 22 months. Because Dr 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 have reported that no potential conflicts of interest exist with any companies/organizations whose products or services may be discussed in this article. Additional information: The e-Appendix can be found in the Supplemental Materials section of the online article.

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]

Somatic Cough Syndrome (Previously Referred to as Psychogenic Cough) and Tic Cough (Previously Referred to as Habit Cough) in Adults and Children: CHEST Guideline and Expert Panel Report.

Somatic cough syndrome or psychogenic cough-what is the difference?

Management of whooping-cough.

Letter: Diagnosis of wheezing and cough.

Defining Chronic Cough: A Systematic Review of the Epidemiological Literature.

Whooping cough: identification, assessment and management.

Chronic dry cough: Diagnostic and management approaches.

The treatment of cough. A comprehensive review.

Cough hypersensitivity syndrome: towards a new approach to chronic cough.

The serological diagnosis of whooping cough.

Chronic Cough.

Whooping cough.

Tic disorders in the differential diagnosis of chronic cough in children in relation to four cases.

Clinicoradiological diagnosis of cough-induced intercostal hernia.

Differential diagnosis of chronic cough in children.

Chronic Cough.

Whooping cough.

Smoking and cough frequency.

The general practice management of whooping cough.

Whooping cough.

Whooping cough.

[Diagnostics and treatment of cough].

Causes of Cough.

Return of whooping cough.