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Unexplained complaints in primary care: Evidence of action bias
ABSTRACT
Purpose Primary care physicians sometimes encounter patients with clinical complaints that do not fit into a recognized diagnostic pattern. This study was undertaken to assess the way physicians respond to patients whose symptoms are unusual or unexplained—that is, what approach they take in the absence of a working hypothesis.
Methods We surveyed 130 primary care physicians affiliated with 3 academic centers in Israel, presenting 5 clinical vignettes describing patients who had unusual complaints, no clear diagnosis, and no apparent need for urgent care. We asked physicians to provide the most likely diagnosis for each case and to rate their level of confidence in that diagnosis;respondents were also asked to provide a management strategy for each case and their level of confidence in the chosen approach. Finally, we asked the physicians to estimate how many of their own patients have presentations similar to the individuals in the clinical vignettes.
Results Physicians proposed, on average, 22 diagnoses for each case. Most indicated that they would choose action (testing, consulting, sending the patient to the emergency department, or prescribing) rather than follow-up only (87% vs 13%; P<.01). Respondents’ confidence in the management approach they had chosen for all the cases was higher than their confidence in the diagnoses (5.6 vs 4.3, respectively, on a scale of1-10; P<.001). Physicians estimated that 10% to 20% of the patients they see in their practice have unusual or unexplained symptoms that are difficult to diagnose.
Conclusion Uncertain diagnosis is a regular challenge for primary care physicians. In such cases, we found that physicians prefer a workup to follow-up, an inclination consistent with“action bias.”
Physicians in primary care sometimes encounter patients with clinical complaints that do not fit into a recognized diagnostic pattern.1 There are varying reports of the prevalence of such cases, ranging from ≤10% when stringent definitions of medically unexplained symptoms are used2 to as high as 40% to 60% of visits.3,4 Unexplained complaints, which may or may not be related to psychiatric disorders, can significantly contribute to high consumption of health care resources.5 Uncertain diagnoses are associated with increased testing6 and false-positive results, which often lead to more tests and complications.7
When physicians face medically unexplained symptoms, their behavior often differs from the watchful waiting approach some recommend.6 This behavior has been attributed to various factors, such as fear of litigation, greater concern about omission than commission, and perception of patient expectations.5 A study involving young patients suggested bias toward intervention for common pediatric diagnoses.8 Using a similar design of physician responses to clinical vignettes,we sought to evaluate a potential bias toward action, such as testing or referral, for patients with unexplained medical complaints.
Methods
Over several months, 2 of us (AK, IG) identified 60 patients in our practices who had(1) unusual medical complaints, (2) no clear diagnosis, and (3) no apparent need for urgent care. After careful consideration, our team selected 5 cases that best fit the above criteria and reflected the widest spectrum of clinical presentations encountered in primary care settings. After removing identifying patient information, we wrote each case up as a clinical vignette, then presented all 5 cases to primary care physicians affiliated with 3 major academic centers. For each case, respondents were asked to provide:
- the most likely diagnosis and their level of confidence in that diagnosis (on a scale of 1 [no confidence] to 10 [complete confidence])
- a management strategy (testing, consulting with a specialist, referral to the emergency department [ED], prescribing medication, or follow-up only) and their level of confidence in that choice.
Physicians were asked to estimate the frequency of such cases in their practice, as well.
Preparation of the data (cleaning, sorting, and filtering) was carried out using JMPv9.0 (SAS Institute, Cary, NC), and analyses were conducted with SPSS v19.0 (IBM,Chicago, Ill). We used descriptive statistics to represent the data and chi-square and ANOVA to compare physicians’ decisions(action vs follow-up). Nonparametric tests were used to compare levels of confidence for diagnosis and management.
Results
We surveyed a convenience sample of 130 primary care physicians affiliated with academic medical centers, 100 of whom responded. Most respondents (62%) were female, and 86% were certified in family medicine. The average age was 45 years (range 30-68 years),with a mean time out of medical school of 17 years (range 1-26 years). Respondents were born in 14 different countries and had undergone medical training in Europe, the United States, or Israel. The diagnoses and management approaches selected for each clinical vignette are presented in TABLE 1. For each case, an average of 22 diagnoses (range 18-25) were proposed. Most physicians (87%; P<.01) indicated that they would choose some type of action (testing, consulting, sending the patient to the ED,or prescribing medication) rather than follow-up alone (TABLE 2). Respondents were able to choose multiple management There appears to be a stronger perceived need to “do something” than to engage in watchful waiting and follow-up.strategies. For all 5 cases, the physicians had more confidence in their patient management approach than in their diagnosis (5.6 vs 4.3;P<.001). On average, men had higher levels of confidence than women for both diagnosis and management (P<.05). Other demographic characteristics, including age, experience, certification, and site of training, were not predictive of confidence level. Respondents estimated that 10% to 20% of their own patients present with unusual and unexplained symptoms, like the patients in the clinical vignettes.
Discussion
Patients with undiagnosed signs and/or symptoms present a significant challenge in primary care. In such cases, physicians prefer a work-up to follow-up, with a confidence level in their management strategy that is higher than for their diagnostic hypotheses. There appears to be a stronger perceived need to “do something” than to engage in watchful waiting and follow-up.
Symptoms subside without treatment. Notably, in all the cases that formed the basis for the clinical vignettes used in our survey, the patients’ complaints eventually subsided, with no specific therapy. In some cases of unclear diagnosis, an active work-up may be justified; in others, watchful waiting before testing for unexplained complaints may be preferable.
Action bias. The preference for action over inaction in all the cases presented suggests what has been described as “action bias.”9 The term is derived from sports; in soccer penalty kicks, for example, it applies to goalkeepers who jump before they can see the kick direction and miss.10 According to the norm theory,11 such errors of commission derive from players’ perception that they are expected to act.10 Conversely, in instances in which inaction is the norm, an omission bias prevails, as people tend to judge acts that are harmful as worse than omissions that are even more harmful.10 In medicine, action bias has been found to influence clinical practice and contribute to overuse of both diagnostic testing and procedures.12-14
Gender difference. Gender has been shown to affect self-perception in cognitive bias.15 In a study of confidence levels among undergraduate students, overconfidence was found to be more prevalent among males than females, particularly for incorrect answers.16 This observation may relate to the gender differences in our study in physicians facing diagnostic uncertainty.
Study limitations. Our research was limited by the nature and type of our sample, but because the inclination to act was found in both immigrant and native practitioners, the observation of action bias could be generalizable to all primary care physicians. The clinical vignettes we chose may not be representative of commonly seen cases of medically unexplained symptoms. Also, our questionnaire was not tested beyond at face validity. It is possible, too, that nonresponders would be less inclined to action in the face of uncertainty. With the high (77%) response rate to our survey, however, their inclusion would be unlikely to strikingly alter the results.
Another limitation inherent to the design of our study is that physicians may respond to vignettes in a way that is substantially different than their response in actual practice. In a practice setting, physicians are able to listen to a full narrative and apply various doctor-patient communication tools, which are especially important in the context of unexplained complaints.17 On the other hand, the artificial setting may reduce the fear of litigation. Our observation of greater confidence in the need for action than for the diagnostic hypothesis is consistent with testing overuse in field studies.6 The fact that our survey went only to physicians affiliated with academic centers is another potential limitation, although it is not clear whether these clinicians differ from nonacademic physicians in their approach to unexplained complaints. Finally, the design of this study did not allow us to explore the reasons for action bias, a task that might be addressed in focus groups or interviews.
A closer look at bias. Our findings suggest a need for more in-depth research on potential biases that drive medical overuse, as part of an overall strategy to improve physicians’approach to medically unexplained symptoms.17 Remedies may require training, practice and failure feedback, quality improvement tools, and innovative management strategies.1,18 Uncertain diagnosis appears to be a frequent challenge in primary care settings. Inthe face of uncertainty, weighing the potential harms of overtesting vs follow-up and facilitating an informed decision-making process with the patient may lead to a reduction inaction bias,19 and thus, in the increased testing and higher health care consumption that often result.
CORRESPONDENCE
Mayer Brezis, MD, Center for Clinical Quality & Safety, Hadassah-Hebrew University Medical Center, 91120 Jerusalem, Israel; brezis@mail.huji.ac.il
ACKNOWLEDGEMENT
The authors thank Steven R. Simon, MD, MPH, for his help with the preparation of this manuscript.
1. Nimnuan C, Hotopf M, Wessely S. Medically unexplained symptoms: an epidemiological study in seven specialities. J Psychosom Res. 2001;51:361-367.
2. Swanson LM, Hamilton JC, Feldman MD. Physician-based estimates of medically unexplained symptoms: a comparison of four case definitions. Fam Pract. 2010;27:487-493.
3. Thomas KB. Temporarily dependent patient in general practice. BMJ. 1974;1:625-626.
4. Jones R, Barraclough K, Dowrick C. When no diagnostic label is applied. BMJ. 2010;340:1302-1304.
5. Todd JW. Wasted resources. Investigations. Lancet. 1984;2:1146-1147.
6. van der Weijden T, van Velsen M, Dinant GJ, et al. Unexplained complaints in general practice: prevalence, patients’ expectations, and professionals’ test-ordering behavior. Med Decis Making. 2003;23:226-231.
7. Brody H. From an ethics of rationing to an ethics of waste avoidance. N Engl J Med. 2012;366:1949-1951.
8. Ayanian JZ, Berwick DM. Do physicians have a bias toward action? A classic study revisited. Med Decis Making. 1991;11:154-158.
9. Patt A, Zeckhauser R. Action bias and environmental decisions. J Risk Uncertain. 2000;21:45-72.
10. Bar-Eli M, Azar OH, Ritov I, et al. Action bias among elite soccer goalkeepers: the case of penalty kicks. J Econ Psychol. 2007;28:606-621.
11. Kahneman D, Miller DT. Norm theory: comparing reality to its alternatives. Psychol Rev. 1986;93:136-153.
12. Doust J, Del Mar C. Why do doctors use treatments that do not work? BMJ. 2004;328:474-475.
13. Scott IA. Errors in clinical reasoning: causes and remedial strategies. BMJ. 2009;338:22-25.
14. Cohain JS. Is action bias one of the numerous causes of ‘unnecaesareans’? Midwif Dig. 2009;19:495-499.
15. Beyer S, Bowden EM. Gender differences in self-perceptions: convergent evidence from three measures of accuracy and bias. Pers Soc Psychol Bull. 1997;23:157-172.
16. Lundeberg MA, Fox PW, Punccohar J. Highly confident but wrong: gender differences and similarities in confidence judgments. J Educ Psychol. 1994;86:114-121.
17. Heijmans M, Olde Hartman TC, van Weel-Baumgarten E, et al. Experts’ opinions on the management of medically unexplained symptoms in primary care. A qualitative analysis of narrative reviews and scientific editorials. Fam Pract. 2011;28:444-455.
18. Croskerry P, Norman G. Overconfidence in clinical decision making. Am J Med. 2008;121(5 suppl):S24-S29.
19. Feinstein AR. The ‘chagrin factor’ and qualitative decision analysis. Arch Intern Med. 1985;145:1257-1259.
Mayer Brezis; MD; MPH; action bias; unexplained complaints; management strategies
ABSTRACT
Purpose Primary care physicians sometimes encounter patients with clinical complaints that do not fit into a recognized diagnostic pattern. This study was undertaken to assess the way physicians respond to patients whose symptoms are unusual or unexplained—that is, what approach they take in the absence of a working hypothesis.
Methods We surveyed 130 primary care physicians affiliated with 3 academic centers in Israel, presenting 5 clinical vignettes describing patients who had unusual complaints, no clear diagnosis, and no apparent need for urgent care. We asked physicians to provide the most likely diagnosis for each case and to rate their level of confidence in that diagnosis;respondents were also asked to provide a management strategy for each case and their level of confidence in the chosen approach. Finally, we asked the physicians to estimate how many of their own patients have presentations similar to the individuals in the clinical vignettes.
Results Physicians proposed, on average, 22 diagnoses for each case. Most indicated that they would choose action (testing, consulting, sending the patient to the emergency department, or prescribing) rather than follow-up only (87% vs 13%; P<.01). Respondents’ confidence in the management approach they had chosen for all the cases was higher than their confidence in the diagnoses (5.6 vs 4.3, respectively, on a scale of1-10; P<.001). Physicians estimated that 10% to 20% of the patients they see in their practice have unusual or unexplained symptoms that are difficult to diagnose.
Conclusion Uncertain diagnosis is a regular challenge for primary care physicians. In such cases, we found that physicians prefer a workup to follow-up, an inclination consistent with“action bias.”
Physicians in primary care sometimes encounter patients with clinical complaints that do not fit into a recognized diagnostic pattern.1 There are varying reports of the prevalence of such cases, ranging from ≤10% when stringent definitions of medically unexplained symptoms are used2 to as high as 40% to 60% of visits.3,4 Unexplained complaints, which may or may not be related to psychiatric disorders, can significantly contribute to high consumption of health care resources.5 Uncertain diagnoses are associated with increased testing6 and false-positive results, which often lead to more tests and complications.7
When physicians face medically unexplained symptoms, their behavior often differs from the watchful waiting approach some recommend.6 This behavior has been attributed to various factors, such as fear of litigation, greater concern about omission than commission, and perception of patient expectations.5 A study involving young patients suggested bias toward intervention for common pediatric diagnoses.8 Using a similar design of physician responses to clinical vignettes,we sought to evaluate a potential bias toward action, such as testing or referral, for patients with unexplained medical complaints.
Methods
Over several months, 2 of us (AK, IG) identified 60 patients in our practices who had(1) unusual medical complaints, (2) no clear diagnosis, and (3) no apparent need for urgent care. After careful consideration, our team selected 5 cases that best fit the above criteria and reflected the widest spectrum of clinical presentations encountered in primary care settings. After removing identifying patient information, we wrote each case up as a clinical vignette, then presented all 5 cases to primary care physicians affiliated with 3 major academic centers. For each case, respondents were asked to provide:
- the most likely diagnosis and their level of confidence in that diagnosis (on a scale of 1 [no confidence] to 10 [complete confidence])
- a management strategy (testing, consulting with a specialist, referral to the emergency department [ED], prescribing medication, or follow-up only) and their level of confidence in that choice.
Physicians were asked to estimate the frequency of such cases in their practice, as well.
Preparation of the data (cleaning, sorting, and filtering) was carried out using JMPv9.0 (SAS Institute, Cary, NC), and analyses were conducted with SPSS v19.0 (IBM,Chicago, Ill). We used descriptive statistics to represent the data and chi-square and ANOVA to compare physicians’ decisions(action vs follow-up). Nonparametric tests were used to compare levels of confidence for diagnosis and management.
Results
We surveyed a convenience sample of 130 primary care physicians affiliated with academic medical centers, 100 of whom responded. Most respondents (62%) were female, and 86% were certified in family medicine. The average age was 45 years (range 30-68 years),with a mean time out of medical school of 17 years (range 1-26 years). Respondents were born in 14 different countries and had undergone medical training in Europe, the United States, or Israel. The diagnoses and management approaches selected for each clinical vignette are presented in TABLE 1. For each case, an average of 22 diagnoses (range 18-25) were proposed. Most physicians (87%; P<.01) indicated that they would choose some type of action (testing, consulting, sending the patient to the ED,or prescribing medication) rather than follow-up alone (TABLE 2). Respondents were able to choose multiple management There appears to be a stronger perceived need to “do something” than to engage in watchful waiting and follow-up.strategies. For all 5 cases, the physicians had more confidence in their patient management approach than in their diagnosis (5.6 vs 4.3;P<.001). On average, men had higher levels of confidence than women for both diagnosis and management (P<.05). Other demographic characteristics, including age, experience, certification, and site of training, were not predictive of confidence level. Respondents estimated that 10% to 20% of their own patients present with unusual and unexplained symptoms, like the patients in the clinical vignettes.
Discussion
Patients with undiagnosed signs and/or symptoms present a significant challenge in primary care. In such cases, physicians prefer a work-up to follow-up, with a confidence level in their management strategy that is higher than for their diagnostic hypotheses. There appears to be a stronger perceived need to “do something” than to engage in watchful waiting and follow-up.
Symptoms subside without treatment. Notably, in all the cases that formed the basis for the clinical vignettes used in our survey, the patients’ complaints eventually subsided, with no specific therapy. In some cases of unclear diagnosis, an active work-up may be justified; in others, watchful waiting before testing for unexplained complaints may be preferable.
Action bias. The preference for action over inaction in all the cases presented suggests what has been described as “action bias.”9 The term is derived from sports; in soccer penalty kicks, for example, it applies to goalkeepers who jump before they can see the kick direction and miss.10 According to the norm theory,11 such errors of commission derive from players’ perception that they are expected to act.10 Conversely, in instances in which inaction is the norm, an omission bias prevails, as people tend to judge acts that are harmful as worse than omissions that are even more harmful.10 In medicine, action bias has been found to influence clinical practice and contribute to overuse of both diagnostic testing and procedures.12-14
Gender difference. Gender has been shown to affect self-perception in cognitive bias.15 In a study of confidence levels among undergraduate students, overconfidence was found to be more prevalent among males than females, particularly for incorrect answers.16 This observation may relate to the gender differences in our study in physicians facing diagnostic uncertainty.
Study limitations. Our research was limited by the nature and type of our sample, but because the inclination to act was found in both immigrant and native practitioners, the observation of action bias could be generalizable to all primary care physicians. The clinical vignettes we chose may not be representative of commonly seen cases of medically unexplained symptoms. Also, our questionnaire was not tested beyond at face validity. It is possible, too, that nonresponders would be less inclined to action in the face of uncertainty. With the high (77%) response rate to our survey, however, their inclusion would be unlikely to strikingly alter the results.
Another limitation inherent to the design of our study is that physicians may respond to vignettes in a way that is substantially different than their response in actual practice. In a practice setting, physicians are able to listen to a full narrative and apply various doctor-patient communication tools, which are especially important in the context of unexplained complaints.17 On the other hand, the artificial setting may reduce the fear of litigation. Our observation of greater confidence in the need for action than for the diagnostic hypothesis is consistent with testing overuse in field studies.6 The fact that our survey went only to physicians affiliated with academic centers is another potential limitation, although it is not clear whether these clinicians differ from nonacademic physicians in their approach to unexplained complaints. Finally, the design of this study did not allow us to explore the reasons for action bias, a task that might be addressed in focus groups or interviews.
A closer look at bias. Our findings suggest a need for more in-depth research on potential biases that drive medical overuse, as part of an overall strategy to improve physicians’approach to medically unexplained symptoms.17 Remedies may require training, practice and failure feedback, quality improvement tools, and innovative management strategies.1,18 Uncertain diagnosis appears to be a frequent challenge in primary care settings. Inthe face of uncertainty, weighing the potential harms of overtesting vs follow-up and facilitating an informed decision-making process with the patient may lead to a reduction inaction bias,19 and thus, in the increased testing and higher health care consumption that often result.
CORRESPONDENCE
Mayer Brezis, MD, Center for Clinical Quality & Safety, Hadassah-Hebrew University Medical Center, 91120 Jerusalem, Israel; brezis@mail.huji.ac.il
ACKNOWLEDGEMENT
The authors thank Steven R. Simon, MD, MPH, for his help with the preparation of this manuscript.
ABSTRACT
Purpose Primary care physicians sometimes encounter patients with clinical complaints that do not fit into a recognized diagnostic pattern. This study was undertaken to assess the way physicians respond to patients whose symptoms are unusual or unexplained—that is, what approach they take in the absence of a working hypothesis.
Methods We surveyed 130 primary care physicians affiliated with 3 academic centers in Israel, presenting 5 clinical vignettes describing patients who had unusual complaints, no clear diagnosis, and no apparent need for urgent care. We asked physicians to provide the most likely diagnosis for each case and to rate their level of confidence in that diagnosis;respondents were also asked to provide a management strategy for each case and their level of confidence in the chosen approach. Finally, we asked the physicians to estimate how many of their own patients have presentations similar to the individuals in the clinical vignettes.
Results Physicians proposed, on average, 22 diagnoses for each case. Most indicated that they would choose action (testing, consulting, sending the patient to the emergency department, or prescribing) rather than follow-up only (87% vs 13%; P<.01). Respondents’ confidence in the management approach they had chosen for all the cases was higher than their confidence in the diagnoses (5.6 vs 4.3, respectively, on a scale of1-10; P<.001). Physicians estimated that 10% to 20% of the patients they see in their practice have unusual or unexplained symptoms that are difficult to diagnose.
Conclusion Uncertain diagnosis is a regular challenge for primary care physicians. In such cases, we found that physicians prefer a workup to follow-up, an inclination consistent with“action bias.”
Physicians in primary care sometimes encounter patients with clinical complaints that do not fit into a recognized diagnostic pattern.1 There are varying reports of the prevalence of such cases, ranging from ≤10% when stringent definitions of medically unexplained symptoms are used2 to as high as 40% to 60% of visits.3,4 Unexplained complaints, which may or may not be related to psychiatric disorders, can significantly contribute to high consumption of health care resources.5 Uncertain diagnoses are associated with increased testing6 and false-positive results, which often lead to more tests and complications.7
When physicians face medically unexplained symptoms, their behavior often differs from the watchful waiting approach some recommend.6 This behavior has been attributed to various factors, such as fear of litigation, greater concern about omission than commission, and perception of patient expectations.5 A study involving young patients suggested bias toward intervention for common pediatric diagnoses.8 Using a similar design of physician responses to clinical vignettes,we sought to evaluate a potential bias toward action, such as testing or referral, for patients with unexplained medical complaints.
Methods
Over several months, 2 of us (AK, IG) identified 60 patients in our practices who had(1) unusual medical complaints, (2) no clear diagnosis, and (3) no apparent need for urgent care. After careful consideration, our team selected 5 cases that best fit the above criteria and reflected the widest spectrum of clinical presentations encountered in primary care settings. After removing identifying patient information, we wrote each case up as a clinical vignette, then presented all 5 cases to primary care physicians affiliated with 3 major academic centers. For each case, respondents were asked to provide:
- the most likely diagnosis and their level of confidence in that diagnosis (on a scale of 1 [no confidence] to 10 [complete confidence])
- a management strategy (testing, consulting with a specialist, referral to the emergency department [ED], prescribing medication, or follow-up only) and their level of confidence in that choice.
Physicians were asked to estimate the frequency of such cases in their practice, as well.
Preparation of the data (cleaning, sorting, and filtering) was carried out using JMPv9.0 (SAS Institute, Cary, NC), and analyses were conducted with SPSS v19.0 (IBM,Chicago, Ill). We used descriptive statistics to represent the data and chi-square and ANOVA to compare physicians’ decisions(action vs follow-up). Nonparametric tests were used to compare levels of confidence for diagnosis and management.
Results
We surveyed a convenience sample of 130 primary care physicians affiliated with academic medical centers, 100 of whom responded. Most respondents (62%) were female, and 86% were certified in family medicine. The average age was 45 years (range 30-68 years),with a mean time out of medical school of 17 years (range 1-26 years). Respondents were born in 14 different countries and had undergone medical training in Europe, the United States, or Israel. The diagnoses and management approaches selected for each clinical vignette are presented in TABLE 1. For each case, an average of 22 diagnoses (range 18-25) were proposed. Most physicians (87%; P<.01) indicated that they would choose some type of action (testing, consulting, sending the patient to the ED,or prescribing medication) rather than follow-up alone (TABLE 2). Respondents were able to choose multiple management There appears to be a stronger perceived need to “do something” than to engage in watchful waiting and follow-up.strategies. For all 5 cases, the physicians had more confidence in their patient management approach than in their diagnosis (5.6 vs 4.3;P<.001). On average, men had higher levels of confidence than women for both diagnosis and management (P<.05). Other demographic characteristics, including age, experience, certification, and site of training, were not predictive of confidence level. Respondents estimated that 10% to 20% of their own patients present with unusual and unexplained symptoms, like the patients in the clinical vignettes.
Discussion
Patients with undiagnosed signs and/or symptoms present a significant challenge in primary care. In such cases, physicians prefer a work-up to follow-up, with a confidence level in their management strategy that is higher than for their diagnostic hypotheses. There appears to be a stronger perceived need to “do something” than to engage in watchful waiting and follow-up.
Symptoms subside without treatment. Notably, in all the cases that formed the basis for the clinical vignettes used in our survey, the patients’ complaints eventually subsided, with no specific therapy. In some cases of unclear diagnosis, an active work-up may be justified; in others, watchful waiting before testing for unexplained complaints may be preferable.
Action bias. The preference for action over inaction in all the cases presented suggests what has been described as “action bias.”9 The term is derived from sports; in soccer penalty kicks, for example, it applies to goalkeepers who jump before they can see the kick direction and miss.10 According to the norm theory,11 such errors of commission derive from players’ perception that they are expected to act.10 Conversely, in instances in which inaction is the norm, an omission bias prevails, as people tend to judge acts that are harmful as worse than omissions that are even more harmful.10 In medicine, action bias has been found to influence clinical practice and contribute to overuse of both diagnostic testing and procedures.12-14
Gender difference. Gender has been shown to affect self-perception in cognitive bias.15 In a study of confidence levels among undergraduate students, overconfidence was found to be more prevalent among males than females, particularly for incorrect answers.16 This observation may relate to the gender differences in our study in physicians facing diagnostic uncertainty.
Study limitations. Our research was limited by the nature and type of our sample, but because the inclination to act was found in both immigrant and native practitioners, the observation of action bias could be generalizable to all primary care physicians. The clinical vignettes we chose may not be representative of commonly seen cases of medically unexplained symptoms. Also, our questionnaire was not tested beyond at face validity. It is possible, too, that nonresponders would be less inclined to action in the face of uncertainty. With the high (77%) response rate to our survey, however, their inclusion would be unlikely to strikingly alter the results.
Another limitation inherent to the design of our study is that physicians may respond to vignettes in a way that is substantially different than their response in actual practice. In a practice setting, physicians are able to listen to a full narrative and apply various doctor-patient communication tools, which are especially important in the context of unexplained complaints.17 On the other hand, the artificial setting may reduce the fear of litigation. Our observation of greater confidence in the need for action than for the diagnostic hypothesis is consistent with testing overuse in field studies.6 The fact that our survey went only to physicians affiliated with academic centers is another potential limitation, although it is not clear whether these clinicians differ from nonacademic physicians in their approach to unexplained complaints. Finally, the design of this study did not allow us to explore the reasons for action bias, a task that might be addressed in focus groups or interviews.
A closer look at bias. Our findings suggest a need for more in-depth research on potential biases that drive medical overuse, as part of an overall strategy to improve physicians’approach to medically unexplained symptoms.17 Remedies may require training, practice and failure feedback, quality improvement tools, and innovative management strategies.1,18 Uncertain diagnosis appears to be a frequent challenge in primary care settings. Inthe face of uncertainty, weighing the potential harms of overtesting vs follow-up and facilitating an informed decision-making process with the patient may lead to a reduction inaction bias,19 and thus, in the increased testing and higher health care consumption that often result.
CORRESPONDENCE
Mayer Brezis, MD, Center for Clinical Quality & Safety, Hadassah-Hebrew University Medical Center, 91120 Jerusalem, Israel; brezis@mail.huji.ac.il
ACKNOWLEDGEMENT
The authors thank Steven R. Simon, MD, MPH, for his help with the preparation of this manuscript.
1. Nimnuan C, Hotopf M, Wessely S. Medically unexplained symptoms: an epidemiological study in seven specialities. J Psychosom Res. 2001;51:361-367.
2. Swanson LM, Hamilton JC, Feldman MD. Physician-based estimates of medically unexplained symptoms: a comparison of four case definitions. Fam Pract. 2010;27:487-493.
3. Thomas KB. Temporarily dependent patient in general practice. BMJ. 1974;1:625-626.
4. Jones R, Barraclough K, Dowrick C. When no diagnostic label is applied. BMJ. 2010;340:1302-1304.
5. Todd JW. Wasted resources. Investigations. Lancet. 1984;2:1146-1147.
6. van der Weijden T, van Velsen M, Dinant GJ, et al. Unexplained complaints in general practice: prevalence, patients’ expectations, and professionals’ test-ordering behavior. Med Decis Making. 2003;23:226-231.
7. Brody H. From an ethics of rationing to an ethics of waste avoidance. N Engl J Med. 2012;366:1949-1951.
8. Ayanian JZ, Berwick DM. Do physicians have a bias toward action? A classic study revisited. Med Decis Making. 1991;11:154-158.
9. Patt A, Zeckhauser R. Action bias and environmental decisions. J Risk Uncertain. 2000;21:45-72.
10. Bar-Eli M, Azar OH, Ritov I, et al. Action bias among elite soccer goalkeepers: the case of penalty kicks. J Econ Psychol. 2007;28:606-621.
11. Kahneman D, Miller DT. Norm theory: comparing reality to its alternatives. Psychol Rev. 1986;93:136-153.
12. Doust J, Del Mar C. Why do doctors use treatments that do not work? BMJ. 2004;328:474-475.
13. Scott IA. Errors in clinical reasoning: causes and remedial strategies. BMJ. 2009;338:22-25.
14. Cohain JS. Is action bias one of the numerous causes of ‘unnecaesareans’? Midwif Dig. 2009;19:495-499.
15. Beyer S, Bowden EM. Gender differences in self-perceptions: convergent evidence from three measures of accuracy and bias. Pers Soc Psychol Bull. 1997;23:157-172.
16. Lundeberg MA, Fox PW, Punccohar J. Highly confident but wrong: gender differences and similarities in confidence judgments. J Educ Psychol. 1994;86:114-121.
17. Heijmans M, Olde Hartman TC, van Weel-Baumgarten E, et al. Experts’ opinions on the management of medically unexplained symptoms in primary care. A qualitative analysis of narrative reviews and scientific editorials. Fam Pract. 2011;28:444-455.
18. Croskerry P, Norman G. Overconfidence in clinical decision making. Am J Med. 2008;121(5 suppl):S24-S29.
19. Feinstein AR. The ‘chagrin factor’ and qualitative decision analysis. Arch Intern Med. 1985;145:1257-1259.
1. Nimnuan C, Hotopf M, Wessely S. Medically unexplained symptoms: an epidemiological study in seven specialities. J Psychosom Res. 2001;51:361-367.
2. Swanson LM, Hamilton JC, Feldman MD. Physician-based estimates of medically unexplained symptoms: a comparison of four case definitions. Fam Pract. 2010;27:487-493.
3. Thomas KB. Temporarily dependent patient in general practice. BMJ. 1974;1:625-626.
4. Jones R, Barraclough K, Dowrick C. When no diagnostic label is applied. BMJ. 2010;340:1302-1304.
5. Todd JW. Wasted resources. Investigations. Lancet. 1984;2:1146-1147.
6. van der Weijden T, van Velsen M, Dinant GJ, et al. Unexplained complaints in general practice: prevalence, patients’ expectations, and professionals’ test-ordering behavior. Med Decis Making. 2003;23:226-231.
7. Brody H. From an ethics of rationing to an ethics of waste avoidance. N Engl J Med. 2012;366:1949-1951.
8. Ayanian JZ, Berwick DM. Do physicians have a bias toward action? A classic study revisited. Med Decis Making. 1991;11:154-158.
9. Patt A, Zeckhauser R. Action bias and environmental decisions. J Risk Uncertain. 2000;21:45-72.
10. Bar-Eli M, Azar OH, Ritov I, et al. Action bias among elite soccer goalkeepers: the case of penalty kicks. J Econ Psychol. 2007;28:606-621.
11. Kahneman D, Miller DT. Norm theory: comparing reality to its alternatives. Psychol Rev. 1986;93:136-153.
12. Doust J, Del Mar C. Why do doctors use treatments that do not work? BMJ. 2004;328:474-475.
13. Scott IA. Errors in clinical reasoning: causes and remedial strategies. BMJ. 2009;338:22-25.
14. Cohain JS. Is action bias one of the numerous causes of ‘unnecaesareans’? Midwif Dig. 2009;19:495-499.
15. Beyer S, Bowden EM. Gender differences in self-perceptions: convergent evidence from three measures of accuracy and bias. Pers Soc Psychol Bull. 1997;23:157-172.
16. Lundeberg MA, Fox PW, Punccohar J. Highly confident but wrong: gender differences and similarities in confidence judgments. J Educ Psychol. 1994;86:114-121.
17. Heijmans M, Olde Hartman TC, van Weel-Baumgarten E, et al. Experts’ opinions on the management of medically unexplained symptoms in primary care. A qualitative analysis of narrative reviews and scientific editorials. Fam Pract. 2011;28:444-455.
18. Croskerry P, Norman G. Overconfidence in clinical decision making. Am J Med. 2008;121(5 suppl):S24-S29.
19. Feinstein AR. The ‘chagrin factor’ and qualitative decision analysis. Arch Intern Med. 1985;145:1257-1259.
Mayer Brezis; MD; MPH; action bias; unexplained complaints; management strategies
Mayer Brezis; MD; MPH; action bias; unexplained complaints; management strategies
Sigh syndrome: Is it a sign of trouble?
- Sigh syndrome is a genuine medical diagnosis with distinct criteria, conferring significant stress for those affected. Despite outward signs of an abnormal breathing pattern, this symptomatology is unrelated to any respiratory or organic pathology.
- Ancillary testing and medication seem unnecessary; supporting reassurance appears sufficient, since the syndrome has a favorable outcome.
Objective The goal of this study was to identify the characteristics and clinical course of patients presenting with considerable stress regarding irrepressible persistent sighing, and to determine whether any association exists between this syndrome and respiratory or other organic disease during the acute or follow-up period.
Study design We conducted a case series review of patients diagnosed with a defined symptom complex and gathered relevant data.
Population Forty patients who presented to 3 clinics in Israel met our 10 criteria for sigh syndrome: recurrent sighing (at least once a minute, for varying lengths of time throughout the day); otherwise shallow respiration; patient conviction that deep breaths are obstructed; intensity of episodes provokes stress leading to consultation; no obvious trigger; episodes last a few days to several weeks; no interference with speech; sighing is absent during sleep; no correlation with physical activity or rest; and self-limited.
Outcomes measured We assessed demographic and health status information, as well as recent circumstances that could have served as triggers for the symptoms. We also performed systematic diagnoses of acute and chronic organic disease.
Results Physicians diagnosed “sigh syndrome” in 40 subjects (19 men [47.5%]), mean age 31.8 years, during the 3-year study period. All patients conformed to 10 sigh syndrome criteria. In 13 patients (32.5%), a significant traumatic event preceded onset of symptoms. Ten (25%) had previous anxiety or somatoform-related disorders. For 23 patients (57.5%), the sigh syndrome episode repeated itself after an initial episode. We found no association in any of the cases with any form of organic disease. Likewise, during the follow-up period (on average, 18 months), we did not observe the development of a specific organic disorder in any case.
Conclusions The “sigh syndrome” runs a benign course; it mainly demands the support and understanding of the treating physician to allay any patient concerns.
In our clinical practices, we have repeatedly cared for patients who came into our clinics because of a worrisome irregular breathing pattern characterized by a deep inspiration, and followed by a noisy expiration. We have referred to the set of clinical signs that these patients present with as “sigh syndrome.”
We have long suspected that sigh syndrome is an underdiagnosed and self-limited condition that is often mistaken for a serious respiratory disorder. In our experience, this syndrome runs a benign course. However, we believed that this syndrome had characteristic and consistent features, and should not be considered a diagnosis of exclusion.
Thus, we undertook a study to observe a group of subjects with these features to judge whether this subjectively alarming symptom complex is in fact harmless, and whether it is appropriate to respond to it as we had, by taking a stress-alleviating approach alone.
What is sigh syndrome?
Patients with sigh syndrome exhibit a compulsion to perform single but repeated deep inspirations, accompanied by a sensation of difficulty in inhaling a sufficient quantity of air. Each inspiration is followed by a prolonged, sometimes noisy expiration—namely, a sigh. Observing such abnormal breaths and confirming that the patient feels a concomitant inability to fill his lungs to capacity is sufficient to make the diagnosis.
This breathing compulsion is irregular in nature: It may occur once a minute or several times a minute, and this breathing pattern may continue—on and off throughout the day—for a few days to several weeks. In our experience, it provokes significant anxiety in patients, prompting them to seek medical advice. It does not occur when the patient is asleep, and it is not triggered by physical activity.
Both patient and doctor may, at first, be convinced that the problem reflects a serious illness. The 10 features of sigh syndrome (TABLE 1) constitute a proposed definition. All of our study subjects exhibited these 10 features.
TABLE 1
10 features of sigh syndrome
|
Sighing as an illness marker
Sighing has been described as one member of a group of signs exhibited by depressed or anxiety-ridden patients.1 While Perin et al2 were the first to point out the importance of distinguishing between sighing and respiratory disease, sighing per se has never been identified as a discrete illness marker.
A number of psychiatric disorders are already well known to incorporate breathing and chest symptoms along with widespread somatization. These include globus hystericus, neurocirculatory asthenia, and Tietze syndrome.3-6 As such, the acute pain of precordial catch syndrome stands out as an example of a distinguishable, clear-cut clinical state devoid of any apparent organic basis.7,8
Methods
How we recruited the patients
Data was collected from 3 family practice clinics in Israel from February 2002 to February 2005. We requested that these practices contribute data of consecutive clinical cases presenting with the 10 set symptoms of sigh syndrome (TABLE 1).
Data collected included basic demographics, the circumstances of the onset of symptoms, concurrent medical conditions, and any associated symptoms. We assessed the patients’ education level by asking questions pertaining to their years of schooling and college degrees. Patients rated their own economic status as being below, average, or above-average income.
The main outcome we examined was the clinical course of the sighing episodes during the ensuing months after their visit, in order to determine whether any patients developed a form of significant organic disease or a disorder that led to hospitalization.
Results
40 cases that cut across the socioeconomic spectrum
Forty patients were recruited for this study. Nineteen (47.5%) were male; their ages ranged from 7 to 53 (mean, 31.8; standard deviation, 13.7). Two patients (5%) were Ashkenazi Jews; 34 (85%) were of North African ethnicity (Sephardic Jews); the other 4 were of varied Asian and European ethnicity. The number of patients with North African ethnicity overrepresents that of the population in their communities. Additionally, 3 members of this group were from the same family.
There was no predilection toward any specific education level or socioeconomic status. The occupations of the subjects were diverse.
Their clinical characteristics
The clinical characteristics of the 40 patients are presented in TABLE 2. The subjects’ sighing began at various times of the day, without relationship to eating or any other activity, and disappeared during sleep. All patients reported the same feeling: that of an extra effort demanded to perform full inspiration. In many cases, adult patients were certain their complaints were a sign of cardiac or respiratory disease, and they were very concerned that there was some grave, underlying disorder.
History and examination failed to reveal evidence of any somatic findings related to breathing difficulty. Breathing rate was within normal limits in all cases.
The physicians’ initial encounters with these patients led to further examination in many cases: electrocardiography, blood oxygen saturation, and complete blood counts. However, no abnormalities were found on any of these tests. Medications, if prescribed at all during these consultations, were usually given to alleviate insomnia or anxiety. Further ancillary investigations or referrals were not ordered.
The sole notable finding on physical examination was a typical murmur (which had already been diagnosed) in a young girl with a congenital atrial septal defect. This 7-year-old developed repeated episodes of sigh syndrome just before her annual visit to the pediatric cardiologist; her mother believed that the child was frightened by the thought of possible future surgery.
TABLE 2
Clinical characteristics of the 40 patients studied—One third experienced a traumatic event
| CHARACTERISTIC | YES | NO |
|---|---|---|
| Smoker | 2 (5%) | 38 (95%) |
| History of anxiety or somatoform disorder | 10 (25%) | 30 (75%) |
| Taking prescription medication at time of diagnosis | 4 (10%) | 36 (90%) |
| Recurrence of sighing episodes during follow-up period | 24 (60%) | 16 (40%) |
| Trigger event (eg, exposure to traumatic event 1 month before presentation with syndrome) | 13 (32.5%) | 27 (67.5%) |
Traumatic events, anxiety disorders suggest stress as a cause
Thirteen patient histories disclosed a definite, recent, significant traumatic event that may have triggered the onset of sigh syndrome. Examples of 2 triggering events are the terrorist murder of several members of a neighbor’s family, and a near-miss with a mortar shell. One subject—a teenager—had recently been left alone in the dark with 2 younger siblings during a power blackout; another young woman said that the sighing episodes began when she decided to get married.
Ten patients had previously diagnosed neurotic disorders, mainly generalized anxiety; these included 2 cases of somatization disorder and 1 case of posttraumatic stress disorder. This information from the patients’ histories was documented in their medical files.
Episodes appear to be self-limited
In all patients, the episodes were self-limited. During the follow-up period, lasting an average of 18 months, none of the patients showed additional medical conditions—respiratory or otherwise—that could be linked to episodes of sigh syndrome. Recurrences of sighing episodes were reported by 24 subjects (60%) after the marker episode. One patient was diagnosed with carcinoma of the pancreas 2 years into follow-up, and later died.
Discussion
A benign, transient disorder
Aside from the solitary and unrelated death noted above, examination and follow-up in all 40 cases did not lead to an alternative diagnosis. Sigh syndrome thus seems to be an entirely benign and transient condition with no sequelae aside from possible recurrences.
Although the pathophysiology is unclear, our finding that 32.5% of patients had a recent traumatic incident strongly suggests a stress-related condition. Furthermore, 25% of the patients were already known to suffer from intermittent anxiety or somatoform disorders, although none were taking medications for these conditions. This adds support to the assumption that a mind-body interaction is underpinning the disorder. Large-scale migration, recent war or terrorist acts, or natural disaster are likely to increase the chances that the average physician will see a patient with sigh syndrome.
A tendency towards North African/Sephardic ethnicity, rather than European ethnicity—in addition to the cluster of 3 cases belonging to the same family—suggests the presentation may be a subconscious cultural, learned, or adopted expression of uneasiness.
Making your evaluation: History and physical are enough
The diagnostic evaluation of sigh syndrome—consisting of careful history-taking and a thorough physical examination—should be sufficient to differentiate it from an array of organic diseases. A physical examination is imperative to exclude other causes for this breathing abnormality.
Ancillary testing is rarely, if ever, indicated. It can perhaps be justified only if the condition is accompanied by an additional (if serendipitous) finding such as the cardiac murmur in the 7-year-old girl noted earlier. Physicians sometimes perform unnecessary investigations, being reluctant to base their diagnoses solely on their clinical expertise.9 A patient may interpret this testing as uncertainty or begin to doubt the diagnosis, thus augmenting—rather than reducing—any anxiety.10 The additional burden of the costs and possible side effects compound the futility of testing indiscriminately.11
Identifying these symptoms with the name “sigh syndrome,” and basing this diagnosis on the history and physical examination, stresses certainty and familiarity with the diagnosis.9 Not only does this reassure the patient, but it eases communication between professionals and forms a basis for research.
Management: Reassure your patient
Management of sigh syndrome consists largely of providing reassurances to your patient. You should emphasize that the condition is real, albeit benign, and that you understand the concern it causes.
Further treatment is unnecessary, aside perhaps from addressing any associated anxiety. A self-limiting (if sometimes recurrent) course can be confidently predicted, and follow-up visits can safely be left to the patient’s own discretion. Since the major correlation with sigh syndrome seems to be stress and the experience of a recent traumatic event, you should always investigate these 2 possibilities when taking the history of a patient with suspected sigh syndrome.
Acknowledgments
This work was inspired and developed by arthur Furst, MD, who died following the submission of this manuscript. Dr Furst was a distinguished and dedicated family physician, a thorough researcher, and a renowned tutor. We dedicate this article to the fond memory of a true leader in the field of rural medicine, and an exceptionally funny and amicable colleague.
Correspondence
Abby Naimer Sody, MD, Gush Katif Health Center, Neve Dekalim, Goosh Katif, Israel 79779; sodyna@clalit.org.il
1. Rakel RE. Textbook of Family Practice. 6th ed. St louis, Mo: WB Saunders; 2002.
2. Perin PV, Perin RJ, Rooklin AR. When a sigh is just a sigh…and not asthma. Ann Allergy 1993;71:478-480.
3. Wilhelm FH, Gevirtz R, Roth WT. Respiratory dys-regulation in anxiety, functional cardiac, and pain disorders. Assessment, phenomenology, and treatment. Behav Modif 2001;25:513-45.
4. Malcomson KG. Globus hystericus vel pharyngis (a reconnaissance of proximal vagal modalities). J Laryngol Otol 1968;82:219-230.
5. Ravich WJ, Wilson RS, Jones B, Donner MW. Psychogenic dysphagia and globus: reevaluation of 23 patients. Dysphagia 1989;4:35-38.
6. Eifert GHB. Cardiophobia: a paradigmatic behavioural model of heart-focused anxiety and non-anginal chest pain. Behav Res Ther 1992;30:329-345.
7. Miller AJ, Texidor TA. “Precordial catch,” a syndrome of anterior chest pain. Ann Intern Med 1959;51:461-467.
8. Sparrow MJ, Bird EL. “Precordial catch”: a benign syndrome of chest pain in young persons. NZ Med J 1978;88:325-326.
9. Balint M. The Doctor, his Patient and the Illness. 2nd ed. London: Sir Isaac Pitman & Sons, 1964.
10. Salmon P. The potentially somatizing effect of clinical consultation. CNS Spectr 2006;11:190-200.
11. Hajioff D, Lowe D. The diagnostic value of barium swallow in globus syndrome. Int J Clin Pract 2004;58:86-89.
- Sigh syndrome is a genuine medical diagnosis with distinct criteria, conferring significant stress for those affected. Despite outward signs of an abnormal breathing pattern, this symptomatology is unrelated to any respiratory or organic pathology.
- Ancillary testing and medication seem unnecessary; supporting reassurance appears sufficient, since the syndrome has a favorable outcome.
Objective The goal of this study was to identify the characteristics and clinical course of patients presenting with considerable stress regarding irrepressible persistent sighing, and to determine whether any association exists between this syndrome and respiratory or other organic disease during the acute or follow-up period.
Study design We conducted a case series review of patients diagnosed with a defined symptom complex and gathered relevant data.
Population Forty patients who presented to 3 clinics in Israel met our 10 criteria for sigh syndrome: recurrent sighing (at least once a minute, for varying lengths of time throughout the day); otherwise shallow respiration; patient conviction that deep breaths are obstructed; intensity of episodes provokes stress leading to consultation; no obvious trigger; episodes last a few days to several weeks; no interference with speech; sighing is absent during sleep; no correlation with physical activity or rest; and self-limited.
Outcomes measured We assessed demographic and health status information, as well as recent circumstances that could have served as triggers for the symptoms. We also performed systematic diagnoses of acute and chronic organic disease.
Results Physicians diagnosed “sigh syndrome” in 40 subjects (19 men [47.5%]), mean age 31.8 years, during the 3-year study period. All patients conformed to 10 sigh syndrome criteria. In 13 patients (32.5%), a significant traumatic event preceded onset of symptoms. Ten (25%) had previous anxiety or somatoform-related disorders. For 23 patients (57.5%), the sigh syndrome episode repeated itself after an initial episode. We found no association in any of the cases with any form of organic disease. Likewise, during the follow-up period (on average, 18 months), we did not observe the development of a specific organic disorder in any case.
Conclusions The “sigh syndrome” runs a benign course; it mainly demands the support and understanding of the treating physician to allay any patient concerns.
In our clinical practices, we have repeatedly cared for patients who came into our clinics because of a worrisome irregular breathing pattern characterized by a deep inspiration, and followed by a noisy expiration. We have referred to the set of clinical signs that these patients present with as “sigh syndrome.”
We have long suspected that sigh syndrome is an underdiagnosed and self-limited condition that is often mistaken for a serious respiratory disorder. In our experience, this syndrome runs a benign course. However, we believed that this syndrome had characteristic and consistent features, and should not be considered a diagnosis of exclusion.
Thus, we undertook a study to observe a group of subjects with these features to judge whether this subjectively alarming symptom complex is in fact harmless, and whether it is appropriate to respond to it as we had, by taking a stress-alleviating approach alone.
What is sigh syndrome?
Patients with sigh syndrome exhibit a compulsion to perform single but repeated deep inspirations, accompanied by a sensation of difficulty in inhaling a sufficient quantity of air. Each inspiration is followed by a prolonged, sometimes noisy expiration—namely, a sigh. Observing such abnormal breaths and confirming that the patient feels a concomitant inability to fill his lungs to capacity is sufficient to make the diagnosis.
This breathing compulsion is irregular in nature: It may occur once a minute or several times a minute, and this breathing pattern may continue—on and off throughout the day—for a few days to several weeks. In our experience, it provokes significant anxiety in patients, prompting them to seek medical advice. It does not occur when the patient is asleep, and it is not triggered by physical activity.
Both patient and doctor may, at first, be convinced that the problem reflects a serious illness. The 10 features of sigh syndrome (TABLE 1) constitute a proposed definition. All of our study subjects exhibited these 10 features.
TABLE 1
10 features of sigh syndrome
|
Sighing as an illness marker
Sighing has been described as one member of a group of signs exhibited by depressed or anxiety-ridden patients.1 While Perin et al2 were the first to point out the importance of distinguishing between sighing and respiratory disease, sighing per se has never been identified as a discrete illness marker.
A number of psychiatric disorders are already well known to incorporate breathing and chest symptoms along with widespread somatization. These include globus hystericus, neurocirculatory asthenia, and Tietze syndrome.3-6 As such, the acute pain of precordial catch syndrome stands out as an example of a distinguishable, clear-cut clinical state devoid of any apparent organic basis.7,8
Methods
How we recruited the patients
Data was collected from 3 family practice clinics in Israel from February 2002 to February 2005. We requested that these practices contribute data of consecutive clinical cases presenting with the 10 set symptoms of sigh syndrome (TABLE 1).
Data collected included basic demographics, the circumstances of the onset of symptoms, concurrent medical conditions, and any associated symptoms. We assessed the patients’ education level by asking questions pertaining to their years of schooling and college degrees. Patients rated their own economic status as being below, average, or above-average income.
The main outcome we examined was the clinical course of the sighing episodes during the ensuing months after their visit, in order to determine whether any patients developed a form of significant organic disease or a disorder that led to hospitalization.
Results
40 cases that cut across the socioeconomic spectrum
Forty patients were recruited for this study. Nineteen (47.5%) were male; their ages ranged from 7 to 53 (mean, 31.8; standard deviation, 13.7). Two patients (5%) were Ashkenazi Jews; 34 (85%) were of North African ethnicity (Sephardic Jews); the other 4 were of varied Asian and European ethnicity. The number of patients with North African ethnicity overrepresents that of the population in their communities. Additionally, 3 members of this group were from the same family.
There was no predilection toward any specific education level or socioeconomic status. The occupations of the subjects were diverse.
Their clinical characteristics
The clinical characteristics of the 40 patients are presented in TABLE 2. The subjects’ sighing began at various times of the day, without relationship to eating or any other activity, and disappeared during sleep. All patients reported the same feeling: that of an extra effort demanded to perform full inspiration. In many cases, adult patients were certain their complaints were a sign of cardiac or respiratory disease, and they were very concerned that there was some grave, underlying disorder.
History and examination failed to reveal evidence of any somatic findings related to breathing difficulty. Breathing rate was within normal limits in all cases.
The physicians’ initial encounters with these patients led to further examination in many cases: electrocardiography, blood oxygen saturation, and complete blood counts. However, no abnormalities were found on any of these tests. Medications, if prescribed at all during these consultations, were usually given to alleviate insomnia or anxiety. Further ancillary investigations or referrals were not ordered.
The sole notable finding on physical examination was a typical murmur (which had already been diagnosed) in a young girl with a congenital atrial septal defect. This 7-year-old developed repeated episodes of sigh syndrome just before her annual visit to the pediatric cardiologist; her mother believed that the child was frightened by the thought of possible future surgery.
TABLE 2
Clinical characteristics of the 40 patients studied—One third experienced a traumatic event
| CHARACTERISTIC | YES | NO |
|---|---|---|
| Smoker | 2 (5%) | 38 (95%) |
| History of anxiety or somatoform disorder | 10 (25%) | 30 (75%) |
| Taking prescription medication at time of diagnosis | 4 (10%) | 36 (90%) |
| Recurrence of sighing episodes during follow-up period | 24 (60%) | 16 (40%) |
| Trigger event (eg, exposure to traumatic event 1 month before presentation with syndrome) | 13 (32.5%) | 27 (67.5%) |
Traumatic events, anxiety disorders suggest stress as a cause
Thirteen patient histories disclosed a definite, recent, significant traumatic event that may have triggered the onset of sigh syndrome. Examples of 2 triggering events are the terrorist murder of several members of a neighbor’s family, and a near-miss with a mortar shell. One subject—a teenager—had recently been left alone in the dark with 2 younger siblings during a power blackout; another young woman said that the sighing episodes began when she decided to get married.
Ten patients had previously diagnosed neurotic disorders, mainly generalized anxiety; these included 2 cases of somatization disorder and 1 case of posttraumatic stress disorder. This information from the patients’ histories was documented in their medical files.
Episodes appear to be self-limited
In all patients, the episodes were self-limited. During the follow-up period, lasting an average of 18 months, none of the patients showed additional medical conditions—respiratory or otherwise—that could be linked to episodes of sigh syndrome. Recurrences of sighing episodes were reported by 24 subjects (60%) after the marker episode. One patient was diagnosed with carcinoma of the pancreas 2 years into follow-up, and later died.
Discussion
A benign, transient disorder
Aside from the solitary and unrelated death noted above, examination and follow-up in all 40 cases did not lead to an alternative diagnosis. Sigh syndrome thus seems to be an entirely benign and transient condition with no sequelae aside from possible recurrences.
Although the pathophysiology is unclear, our finding that 32.5% of patients had a recent traumatic incident strongly suggests a stress-related condition. Furthermore, 25% of the patients were already known to suffer from intermittent anxiety or somatoform disorders, although none were taking medications for these conditions. This adds support to the assumption that a mind-body interaction is underpinning the disorder. Large-scale migration, recent war or terrorist acts, or natural disaster are likely to increase the chances that the average physician will see a patient with sigh syndrome.
A tendency towards North African/Sephardic ethnicity, rather than European ethnicity—in addition to the cluster of 3 cases belonging to the same family—suggests the presentation may be a subconscious cultural, learned, or adopted expression of uneasiness.
Making your evaluation: History and physical are enough
The diagnostic evaluation of sigh syndrome—consisting of careful history-taking and a thorough physical examination—should be sufficient to differentiate it from an array of organic diseases. A physical examination is imperative to exclude other causes for this breathing abnormality.
Ancillary testing is rarely, if ever, indicated. It can perhaps be justified only if the condition is accompanied by an additional (if serendipitous) finding such as the cardiac murmur in the 7-year-old girl noted earlier. Physicians sometimes perform unnecessary investigations, being reluctant to base their diagnoses solely on their clinical expertise.9 A patient may interpret this testing as uncertainty or begin to doubt the diagnosis, thus augmenting—rather than reducing—any anxiety.10 The additional burden of the costs and possible side effects compound the futility of testing indiscriminately.11
Identifying these symptoms with the name “sigh syndrome,” and basing this diagnosis on the history and physical examination, stresses certainty and familiarity with the diagnosis.9 Not only does this reassure the patient, but it eases communication between professionals and forms a basis for research.
Management: Reassure your patient
Management of sigh syndrome consists largely of providing reassurances to your patient. You should emphasize that the condition is real, albeit benign, and that you understand the concern it causes.
Further treatment is unnecessary, aside perhaps from addressing any associated anxiety. A self-limiting (if sometimes recurrent) course can be confidently predicted, and follow-up visits can safely be left to the patient’s own discretion. Since the major correlation with sigh syndrome seems to be stress and the experience of a recent traumatic event, you should always investigate these 2 possibilities when taking the history of a patient with suspected sigh syndrome.
Acknowledgments
This work was inspired and developed by arthur Furst, MD, who died following the submission of this manuscript. Dr Furst was a distinguished and dedicated family physician, a thorough researcher, and a renowned tutor. We dedicate this article to the fond memory of a true leader in the field of rural medicine, and an exceptionally funny and amicable colleague.
Correspondence
Abby Naimer Sody, MD, Gush Katif Health Center, Neve Dekalim, Goosh Katif, Israel 79779; sodyna@clalit.org.il
- Sigh syndrome is a genuine medical diagnosis with distinct criteria, conferring significant stress for those affected. Despite outward signs of an abnormal breathing pattern, this symptomatology is unrelated to any respiratory or organic pathology.
- Ancillary testing and medication seem unnecessary; supporting reassurance appears sufficient, since the syndrome has a favorable outcome.
Objective The goal of this study was to identify the characteristics and clinical course of patients presenting with considerable stress regarding irrepressible persistent sighing, and to determine whether any association exists between this syndrome and respiratory or other organic disease during the acute or follow-up period.
Study design We conducted a case series review of patients diagnosed with a defined symptom complex and gathered relevant data.
Population Forty patients who presented to 3 clinics in Israel met our 10 criteria for sigh syndrome: recurrent sighing (at least once a minute, for varying lengths of time throughout the day); otherwise shallow respiration; patient conviction that deep breaths are obstructed; intensity of episodes provokes stress leading to consultation; no obvious trigger; episodes last a few days to several weeks; no interference with speech; sighing is absent during sleep; no correlation with physical activity or rest; and self-limited.
Outcomes measured We assessed demographic and health status information, as well as recent circumstances that could have served as triggers for the symptoms. We also performed systematic diagnoses of acute and chronic organic disease.
Results Physicians diagnosed “sigh syndrome” in 40 subjects (19 men [47.5%]), mean age 31.8 years, during the 3-year study period. All patients conformed to 10 sigh syndrome criteria. In 13 patients (32.5%), a significant traumatic event preceded onset of symptoms. Ten (25%) had previous anxiety or somatoform-related disorders. For 23 patients (57.5%), the sigh syndrome episode repeated itself after an initial episode. We found no association in any of the cases with any form of organic disease. Likewise, during the follow-up period (on average, 18 months), we did not observe the development of a specific organic disorder in any case.
Conclusions The “sigh syndrome” runs a benign course; it mainly demands the support and understanding of the treating physician to allay any patient concerns.
In our clinical practices, we have repeatedly cared for patients who came into our clinics because of a worrisome irregular breathing pattern characterized by a deep inspiration, and followed by a noisy expiration. We have referred to the set of clinical signs that these patients present with as “sigh syndrome.”
We have long suspected that sigh syndrome is an underdiagnosed and self-limited condition that is often mistaken for a serious respiratory disorder. In our experience, this syndrome runs a benign course. However, we believed that this syndrome had characteristic and consistent features, and should not be considered a diagnosis of exclusion.
Thus, we undertook a study to observe a group of subjects with these features to judge whether this subjectively alarming symptom complex is in fact harmless, and whether it is appropriate to respond to it as we had, by taking a stress-alleviating approach alone.
What is sigh syndrome?
Patients with sigh syndrome exhibit a compulsion to perform single but repeated deep inspirations, accompanied by a sensation of difficulty in inhaling a sufficient quantity of air. Each inspiration is followed by a prolonged, sometimes noisy expiration—namely, a sigh. Observing such abnormal breaths and confirming that the patient feels a concomitant inability to fill his lungs to capacity is sufficient to make the diagnosis.
This breathing compulsion is irregular in nature: It may occur once a minute or several times a minute, and this breathing pattern may continue—on and off throughout the day—for a few days to several weeks. In our experience, it provokes significant anxiety in patients, prompting them to seek medical advice. It does not occur when the patient is asleep, and it is not triggered by physical activity.
Both patient and doctor may, at first, be convinced that the problem reflects a serious illness. The 10 features of sigh syndrome (TABLE 1) constitute a proposed definition. All of our study subjects exhibited these 10 features.
TABLE 1
10 features of sigh syndrome
|
Sighing as an illness marker
Sighing has been described as one member of a group of signs exhibited by depressed or anxiety-ridden patients.1 While Perin et al2 were the first to point out the importance of distinguishing between sighing and respiratory disease, sighing per se has never been identified as a discrete illness marker.
A number of psychiatric disorders are already well known to incorporate breathing and chest symptoms along with widespread somatization. These include globus hystericus, neurocirculatory asthenia, and Tietze syndrome.3-6 As such, the acute pain of precordial catch syndrome stands out as an example of a distinguishable, clear-cut clinical state devoid of any apparent organic basis.7,8
Methods
How we recruited the patients
Data was collected from 3 family practice clinics in Israel from February 2002 to February 2005. We requested that these practices contribute data of consecutive clinical cases presenting with the 10 set symptoms of sigh syndrome (TABLE 1).
Data collected included basic demographics, the circumstances of the onset of symptoms, concurrent medical conditions, and any associated symptoms. We assessed the patients’ education level by asking questions pertaining to their years of schooling and college degrees. Patients rated their own economic status as being below, average, or above-average income.
The main outcome we examined was the clinical course of the sighing episodes during the ensuing months after their visit, in order to determine whether any patients developed a form of significant organic disease or a disorder that led to hospitalization.
Results
40 cases that cut across the socioeconomic spectrum
Forty patients were recruited for this study. Nineteen (47.5%) were male; their ages ranged from 7 to 53 (mean, 31.8; standard deviation, 13.7). Two patients (5%) were Ashkenazi Jews; 34 (85%) were of North African ethnicity (Sephardic Jews); the other 4 were of varied Asian and European ethnicity. The number of patients with North African ethnicity overrepresents that of the population in their communities. Additionally, 3 members of this group were from the same family.
There was no predilection toward any specific education level or socioeconomic status. The occupations of the subjects were diverse.
Their clinical characteristics
The clinical characteristics of the 40 patients are presented in TABLE 2. The subjects’ sighing began at various times of the day, without relationship to eating or any other activity, and disappeared during sleep. All patients reported the same feeling: that of an extra effort demanded to perform full inspiration. In many cases, adult patients were certain their complaints were a sign of cardiac or respiratory disease, and they were very concerned that there was some grave, underlying disorder.
History and examination failed to reveal evidence of any somatic findings related to breathing difficulty. Breathing rate was within normal limits in all cases.
The physicians’ initial encounters with these patients led to further examination in many cases: electrocardiography, blood oxygen saturation, and complete blood counts. However, no abnormalities were found on any of these tests. Medications, if prescribed at all during these consultations, were usually given to alleviate insomnia or anxiety. Further ancillary investigations or referrals were not ordered.
The sole notable finding on physical examination was a typical murmur (which had already been diagnosed) in a young girl with a congenital atrial septal defect. This 7-year-old developed repeated episodes of sigh syndrome just before her annual visit to the pediatric cardiologist; her mother believed that the child was frightened by the thought of possible future surgery.
TABLE 2
Clinical characteristics of the 40 patients studied—One third experienced a traumatic event
| CHARACTERISTIC | YES | NO |
|---|---|---|
| Smoker | 2 (5%) | 38 (95%) |
| History of anxiety or somatoform disorder | 10 (25%) | 30 (75%) |
| Taking prescription medication at time of diagnosis | 4 (10%) | 36 (90%) |
| Recurrence of sighing episodes during follow-up period | 24 (60%) | 16 (40%) |
| Trigger event (eg, exposure to traumatic event 1 month before presentation with syndrome) | 13 (32.5%) | 27 (67.5%) |
Traumatic events, anxiety disorders suggest stress as a cause
Thirteen patient histories disclosed a definite, recent, significant traumatic event that may have triggered the onset of sigh syndrome. Examples of 2 triggering events are the terrorist murder of several members of a neighbor’s family, and a near-miss with a mortar shell. One subject—a teenager—had recently been left alone in the dark with 2 younger siblings during a power blackout; another young woman said that the sighing episodes began when she decided to get married.
Ten patients had previously diagnosed neurotic disorders, mainly generalized anxiety; these included 2 cases of somatization disorder and 1 case of posttraumatic stress disorder. This information from the patients’ histories was documented in their medical files.
Episodes appear to be self-limited
In all patients, the episodes were self-limited. During the follow-up period, lasting an average of 18 months, none of the patients showed additional medical conditions—respiratory or otherwise—that could be linked to episodes of sigh syndrome. Recurrences of sighing episodes were reported by 24 subjects (60%) after the marker episode. One patient was diagnosed with carcinoma of the pancreas 2 years into follow-up, and later died.
Discussion
A benign, transient disorder
Aside from the solitary and unrelated death noted above, examination and follow-up in all 40 cases did not lead to an alternative diagnosis. Sigh syndrome thus seems to be an entirely benign and transient condition with no sequelae aside from possible recurrences.
Although the pathophysiology is unclear, our finding that 32.5% of patients had a recent traumatic incident strongly suggests a stress-related condition. Furthermore, 25% of the patients were already known to suffer from intermittent anxiety or somatoform disorders, although none were taking medications for these conditions. This adds support to the assumption that a mind-body interaction is underpinning the disorder. Large-scale migration, recent war or terrorist acts, or natural disaster are likely to increase the chances that the average physician will see a patient with sigh syndrome.
A tendency towards North African/Sephardic ethnicity, rather than European ethnicity—in addition to the cluster of 3 cases belonging to the same family—suggests the presentation may be a subconscious cultural, learned, or adopted expression of uneasiness.
Making your evaluation: History and physical are enough
The diagnostic evaluation of sigh syndrome—consisting of careful history-taking and a thorough physical examination—should be sufficient to differentiate it from an array of organic diseases. A physical examination is imperative to exclude other causes for this breathing abnormality.
Ancillary testing is rarely, if ever, indicated. It can perhaps be justified only if the condition is accompanied by an additional (if serendipitous) finding such as the cardiac murmur in the 7-year-old girl noted earlier. Physicians sometimes perform unnecessary investigations, being reluctant to base their diagnoses solely on their clinical expertise.9 A patient may interpret this testing as uncertainty or begin to doubt the diagnosis, thus augmenting—rather than reducing—any anxiety.10 The additional burden of the costs and possible side effects compound the futility of testing indiscriminately.11
Identifying these symptoms with the name “sigh syndrome,” and basing this diagnosis on the history and physical examination, stresses certainty and familiarity with the diagnosis.9 Not only does this reassure the patient, but it eases communication between professionals and forms a basis for research.
Management: Reassure your patient
Management of sigh syndrome consists largely of providing reassurances to your patient. You should emphasize that the condition is real, albeit benign, and that you understand the concern it causes.
Further treatment is unnecessary, aside perhaps from addressing any associated anxiety. A self-limiting (if sometimes recurrent) course can be confidently predicted, and follow-up visits can safely be left to the patient’s own discretion. Since the major correlation with sigh syndrome seems to be stress and the experience of a recent traumatic event, you should always investigate these 2 possibilities when taking the history of a patient with suspected sigh syndrome.
Acknowledgments
This work was inspired and developed by arthur Furst, MD, who died following the submission of this manuscript. Dr Furst was a distinguished and dedicated family physician, a thorough researcher, and a renowned tutor. We dedicate this article to the fond memory of a true leader in the field of rural medicine, and an exceptionally funny and amicable colleague.
Correspondence
Abby Naimer Sody, MD, Gush Katif Health Center, Neve Dekalim, Goosh Katif, Israel 79779; sodyna@clalit.org.il
1. Rakel RE. Textbook of Family Practice. 6th ed. St louis, Mo: WB Saunders; 2002.
2. Perin PV, Perin RJ, Rooklin AR. When a sigh is just a sigh…and not asthma. Ann Allergy 1993;71:478-480.
3. Wilhelm FH, Gevirtz R, Roth WT. Respiratory dys-regulation in anxiety, functional cardiac, and pain disorders. Assessment, phenomenology, and treatment. Behav Modif 2001;25:513-45.
4. Malcomson KG. Globus hystericus vel pharyngis (a reconnaissance of proximal vagal modalities). J Laryngol Otol 1968;82:219-230.
5. Ravich WJ, Wilson RS, Jones B, Donner MW. Psychogenic dysphagia and globus: reevaluation of 23 patients. Dysphagia 1989;4:35-38.
6. Eifert GHB. Cardiophobia: a paradigmatic behavioural model of heart-focused anxiety and non-anginal chest pain. Behav Res Ther 1992;30:329-345.
7. Miller AJ, Texidor TA. “Precordial catch,” a syndrome of anterior chest pain. Ann Intern Med 1959;51:461-467.
8. Sparrow MJ, Bird EL. “Precordial catch”: a benign syndrome of chest pain in young persons. NZ Med J 1978;88:325-326.
9. Balint M. The Doctor, his Patient and the Illness. 2nd ed. London: Sir Isaac Pitman & Sons, 1964.
10. Salmon P. The potentially somatizing effect of clinical consultation. CNS Spectr 2006;11:190-200.
11. Hajioff D, Lowe D. The diagnostic value of barium swallow in globus syndrome. Int J Clin Pract 2004;58:86-89.
1. Rakel RE. Textbook of Family Practice. 6th ed. St louis, Mo: WB Saunders; 2002.
2. Perin PV, Perin RJ, Rooklin AR. When a sigh is just a sigh…and not asthma. Ann Allergy 1993;71:478-480.
3. Wilhelm FH, Gevirtz R, Roth WT. Respiratory dys-regulation in anxiety, functional cardiac, and pain disorders. Assessment, phenomenology, and treatment. Behav Modif 2001;25:513-45.
4. Malcomson KG. Globus hystericus vel pharyngis (a reconnaissance of proximal vagal modalities). J Laryngol Otol 1968;82:219-230.
5. Ravich WJ, Wilson RS, Jones B, Donner MW. Psychogenic dysphagia and globus: reevaluation of 23 patients. Dysphagia 1989;4:35-38.
6. Eifert GHB. Cardiophobia: a paradigmatic behavioural model of heart-focused anxiety and non-anginal chest pain. Behav Res Ther 1992;30:329-345.
7. Miller AJ, Texidor TA. “Precordial catch,” a syndrome of anterior chest pain. Ann Intern Med 1959;51:461-467.
8. Sparrow MJ, Bird EL. “Precordial catch”: a benign syndrome of chest pain in young persons. NZ Med J 1978;88:325-326.
9. Balint M. The Doctor, his Patient and the Illness. 2nd ed. London: Sir Isaac Pitman & Sons, 1964.
10. Salmon P. The potentially somatizing effect of clinical consultation. CNS Spectr 2006;11:190-200.
11. Hajioff D, Lowe D. The diagnostic value of barium swallow in globus syndrome. Int J Clin Pract 2004;58:86-89.