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Treating empty nose syndrome as a somatic symptom disorder
General Hospital Psychiatry 37 (2015) 273.e9–273.e10
Contents lists available at ScienceDirect
General Hospital Psychiatry journal homepage: http://www.ghpjournal.com
Case Report
Treating empty nose syndrome as a somatic symptom disorder Cédric Lemogne, M.D., Ph.D. a,b,c,⁎, Silla M. Consoli, M.D., Ph.D. a,b, Frédéric Limosin, M.D., Ph.D. a,b,c, Pierre Bonfils, M.D., Ph.D. a,d,e a
Université Paris Descartes, Sorbonne Paris Cité, Faculté de Médecine, Paris, France AP-HP, Hôpitaux Universitaires Paris Ouest, Service de Psychiatrie de l'Adulte et du Sujet Âgé, Paris, France Inserm, U894, Centre Psychiatrie et Neurosciences, Paris, France d AP-HP, Hôpitaux Universitaires Paris Ouest, Service d'ORL et de Chirurgie Cervico-Faciale, Paris, France e CNRS-SSA UMR MD8257, Cognition and Action Group (Cognac G), Paris, France b c
a r t i c l e
i n f o
Article history: Received 6 January 2015 Revised 13 February 2015 Accepted 17 February 2015 Keywords: Treatment Empty nose syndrome Functional somatic syndromes Somatic symptom disorders Somatoform disorders
a b s t r a c t Empty nose syndrome (ENS) is a rare complication of inferior turbinate resection, characterized by a paradoxical nasal obstruction sensation despite decreased nasal resistance. Here we report the case of a 37-year-old patient with ENS and severe functional impairment, who was diagnosed with a somatic symptom disorder and treated accordingly. Cognitive behavior therapy targeting dysfunctional beliefs and avoidance behaviors together with a treatment by venlafaxine resulted in dramatic functional improvement between month 2 and month 4. At month 6, the patient was displaying back-to-normal levels of functioning and was no longer seeking care for ENS. Treating ENS as a somatic symptom disorder might constitute a first-line, safe alternative to surgical treatment. © 2015 Elsevier Inc. All rights reserved.
Empty nose syndrome (ENS), a term first coined by Kern and Stenkvist in 1994, is a rare yet potentially debilitating complication of inferior turbinate resection [1,2]. It is characterized by a paradoxical nasal obstruction sensation despite decreased nasal resistance. Others symptoms typically include nasal dryness, dyspnea, hyposmia, sleep disorders, fatigue, anxiety and depression. The ENS incidence is unknown but estimates range up to 20% [1]. Conservative medical management mainly relies on aggressive nasal hygiene and nasal moisturizers. Although the ENS pathogenesis remains unknown [2], it is sometimes assumed to merely result from anatomical changes, leading to proposals of nasal augmentation for refractory cases [3]. Here we report the case of a 37-year-old patient with ENS, which was treated as a somatic symptom disorder. This married, socially integrated tree pruner with two children underwent an inferior turbinate resection in February 2012 for nasal obstruction due to a nonallergic chronic rhinitis. Thereafter, he had suffered from gradually increasing ENS symptoms with severe functional impairment. At the first consultation (May 2013), he was no longer working, had left almost all social relationships and leisure activities, was spending most of his time confined in his bedroom, with a sophisticated air humidifier. As recommended by several physicians, he was ⁎ Corresponding author. Unité de Psychologie et Psychiatrie de Liaison et D'urgence, Hôpital Européen Georges Pompidou, 20 rue Leblanc, 75908 Paris Cedex 15, France. Tel.: +33-1-56-09-33-71; fax: +33-1-56-09-31-46. E-mail address: [email protected] (C. Lemogne). http://dx.doi.org/10.1016/j.genhosppsych.2015.02.005 0163-8343/© 2015 Elsevier Inc. All rights reserved.
performing nasal irrigations several times per day but refused proposals of nasal augmentation. A polysomnography had found no support for sleep apnea. According to the frequent comorbidity with anxiety and depression, we decided to offer patients with ENS joint consultations with an otorhinolaryngologist and a consultation-liaison psychiatrist. Contrasting with the patient's expectation, the physical examination by the otorhinolaryngologist (P.B.) was merely normal. This poor correlation between subjective and objective findings is a distinctive feature of the ENS [1]. Indeed, from the psychiatrist's perspective (C.L.), the patient was presenting with a typical somatic symptom disorder with comorbid depression and symptoms of panic attacks. Depressive symptoms included a loss of interest and pleasure in almost all activities, persistent low mood, pessimism, difficulties concentrating, sleep disorders and fatigue. Symptoms of panic attacks were sudden respiratory symptoms including shortness of breath and choking sensations associated with catastrophizing and fear of impending death. Such excessive and disproportionate thoughts associated with somatic symptoms are one central feature of somatic symptom disorders. These disorders, frequently referred to as “functional somatic syndromes,” used to be negatively defined by “medically unexplained symptoms.” Compared with the older category of “somatoform disorders,” the construct of “somatic symptoms disorders” more accurately acknowledges that these symptoms may or may not be associated with organic lesions. The rarity of the ENS after inferior turbinate resection suggests that anatomical changes may not fully account for the symptoms and their functional
273.e10
C. Lemogne et al. / General Hospital Psychiatry 37 (2015) 273.e9–273.e10
impact [2]. Functional brain imaging rather suggests that ENS may share some psychophysiological mechanisms, including increased limbic reactivity, with other functional somatic syndromes such as fibromyalgia and irritable bowel syndrome [4,5]. Diagnosing the patient with a somatic symptom disorder was helpful in implementing efficient treatment, based on four principles: building and maintaining therapeutic alliance despite initial skepticism; targeting core dysfunctional beliefs (e.g., that ENS symptoms are predominantly caused by anatomical changes) with cognitive therapy; eradicating avoidance behaviors (i.e., those aiming at decreasing ENS symptoms) with behavioral therapy; prescribing a serotonin reuptake inhibitor, even in the absence of comorbid anxiety or depression. This strategy is efficient in treating fibromyalgia [6] or irritable bowel syndrome [7]. None of these interventions had been proposed to the patient until he was diagnosed with a somatic symptom disorder. Although therapeutic alliance is warranted in any patient–doctor relationship [8], it is especially challenging to achieve with patients with somatic symptom disorders who are typically reluctant to any psychological account of their symptoms. Because of the painful nature of the symptoms, a serotonin and norepinephrine reuptake inhibitor was preferred and introduced at low dosage (i.e., venlafaxine 37.5 mg). The patient progressively stepped back from dysfunctional beliefs through standard cognitive therapy techniques (e.g., socratic questioning) while becoming able to identify and leave several avoidance behaviors. These behaviors were nasal irrigations and the use of an air humidifier (aiming at reducing nasal dryness), repeated medical consultations (aiming at reducing the fear of having an undiagnosed hazardous physical condition) and physical inactivity (aiming at reducing dyspnea). Venlafaxine was gradually increased up to 150 mg over 3 months, with weight gain and a transient increase of nose dryness as side effects. Although the patient did not report much improvement of ENS symptoms per se, dramatic functional improvement was observed between month 2 and month 4 and confirmed by patients' relatives. At month 6, he was working again, displaying back-to-normal levels of functioning in social and leisure activities and was no longer seeking care for ENS. At month 12, venlafaxine was gradually stopped, without functional relapse at month 18.
Although many clinicians emphasize the potential role of psychological mechanisms in the ENS etiology and impact, targeting these mechanisms is rarely considered as central. Although we cannot determine the respective weight of cognitive behavior therapy and venlafaxine in the remediation of this patient, this case report suggests that treating ENS as a somatic symptom disorder might constitute a first-line, safe alternative to surgical or even medical treatment. It is noteworthy that eradicating avoidance behaviors may conflict with usual recommendations (e.g., nasal irrigations) but is critical as they may maintain ENS symptoms through operant conditioning. Future studies are warranted to establish the efficacy and safety of this approach.
Acknowledgments Conflicts of interest: None. Funding source: None.
References [1] Chhabra N, Houser SM. The diagnosis and management of empty nose syndrome. Otolaryngol Clin North Am 2009;42:311–30. [2] Sozansky J, Houser SM. Pathophysiology of empty nose syndrome. Laryngoscope 2015;125:70–4. [3] Jiang C, Wong F, Chen K, Shi R. Assessment of surgical results in patients with empty nose syndrome using the 25-item Sino-Nasal Outcome Test Evaluation. JAMA Otolaryngol Head Neck Surg 2014;140:453–8. [4] Browning M, Fletcher P, Sharpe M. Can neuroimaging help us to understand and classify somatoform disorders? A systematic and critical review. Psychosom Med 2011; 73:173–84. [5] Freund W, Wunderlich AP, Stöcker T, Schmitz BL, Scheithauer MO. Empty nose syndrome: limbic system activation observed by functional magnetic resonance imaging. Laryngoscope 2011;121:2019–25. [6] Clauw DJ. Fibromyalgia: a clinical review. JAMA 2014;311:1547–55. [7] Ford AC, Quigley EMM, Lacy BE, Lembo AJ, Saito YA, Schiller LR, et al. Effect of antidepressants and psychological therapies, including hypnotherapy, in irritable bowel syndrome: systematic review and meta-analysis. Am J Gastroenterol 2014;109: 1350–65. [8] Attale C, Lemogne C, Sola-Gazagnes A, Guedeney N, Slama G, Horvath A-O, et al. Therapeutic alliance and glycaemic control in type 1 diabetes: a pilot study. Diabetes Metab 2010;36:499–502.
Contents lists available at ScienceDirect
General Hospital Psychiatry journal homepage: http://www.ghpjournal.com
Case Report
Treating empty nose syndrome as a somatic symptom disorder Cédric Lemogne, M.D., Ph.D. a,b,c,⁎, Silla M. Consoli, M.D., Ph.D. a,b, Frédéric Limosin, M.D., Ph.D. a,b,c, Pierre Bonfils, M.D., Ph.D. a,d,e a
Université Paris Descartes, Sorbonne Paris Cité, Faculté de Médecine, Paris, France AP-HP, Hôpitaux Universitaires Paris Ouest, Service de Psychiatrie de l'Adulte et du Sujet Âgé, Paris, France Inserm, U894, Centre Psychiatrie et Neurosciences, Paris, France d AP-HP, Hôpitaux Universitaires Paris Ouest, Service d'ORL et de Chirurgie Cervico-Faciale, Paris, France e CNRS-SSA UMR MD8257, Cognition and Action Group (Cognac G), Paris, France b c
a r t i c l e
i n f o
Article history: Received 6 January 2015 Revised 13 February 2015 Accepted 17 February 2015 Keywords: Treatment Empty nose syndrome Functional somatic syndromes Somatic symptom disorders Somatoform disorders
a b s t r a c t Empty nose syndrome (ENS) is a rare complication of inferior turbinate resection, characterized by a paradoxical nasal obstruction sensation despite decreased nasal resistance. Here we report the case of a 37-year-old patient with ENS and severe functional impairment, who was diagnosed with a somatic symptom disorder and treated accordingly. Cognitive behavior therapy targeting dysfunctional beliefs and avoidance behaviors together with a treatment by venlafaxine resulted in dramatic functional improvement between month 2 and month 4. At month 6, the patient was displaying back-to-normal levels of functioning and was no longer seeking care for ENS. Treating ENS as a somatic symptom disorder might constitute a first-line, safe alternative to surgical treatment. © 2015 Elsevier Inc. All rights reserved.
Empty nose syndrome (ENS), a term first coined by Kern and Stenkvist in 1994, is a rare yet potentially debilitating complication of inferior turbinate resection [1,2]. It is characterized by a paradoxical nasal obstruction sensation despite decreased nasal resistance. Others symptoms typically include nasal dryness, dyspnea, hyposmia, sleep disorders, fatigue, anxiety and depression. The ENS incidence is unknown but estimates range up to 20% [1]. Conservative medical management mainly relies on aggressive nasal hygiene and nasal moisturizers. Although the ENS pathogenesis remains unknown [2], it is sometimes assumed to merely result from anatomical changes, leading to proposals of nasal augmentation for refractory cases [3]. Here we report the case of a 37-year-old patient with ENS, which was treated as a somatic symptom disorder. This married, socially integrated tree pruner with two children underwent an inferior turbinate resection in February 2012 for nasal obstruction due to a nonallergic chronic rhinitis. Thereafter, he had suffered from gradually increasing ENS symptoms with severe functional impairment. At the first consultation (May 2013), he was no longer working, had left almost all social relationships and leisure activities, was spending most of his time confined in his bedroom, with a sophisticated air humidifier. As recommended by several physicians, he was ⁎ Corresponding author. Unité de Psychologie et Psychiatrie de Liaison et D'urgence, Hôpital Européen Georges Pompidou, 20 rue Leblanc, 75908 Paris Cedex 15, France. Tel.: +33-1-56-09-33-71; fax: +33-1-56-09-31-46. E-mail address: [email protected] (C. Lemogne). http://dx.doi.org/10.1016/j.genhosppsych.2015.02.005 0163-8343/© 2015 Elsevier Inc. All rights reserved.
performing nasal irrigations several times per day but refused proposals of nasal augmentation. A polysomnography had found no support for sleep apnea. According to the frequent comorbidity with anxiety and depression, we decided to offer patients with ENS joint consultations with an otorhinolaryngologist and a consultation-liaison psychiatrist. Contrasting with the patient's expectation, the physical examination by the otorhinolaryngologist (P.B.) was merely normal. This poor correlation between subjective and objective findings is a distinctive feature of the ENS [1]. Indeed, from the psychiatrist's perspective (C.L.), the patient was presenting with a typical somatic symptom disorder with comorbid depression and symptoms of panic attacks. Depressive symptoms included a loss of interest and pleasure in almost all activities, persistent low mood, pessimism, difficulties concentrating, sleep disorders and fatigue. Symptoms of panic attacks were sudden respiratory symptoms including shortness of breath and choking sensations associated with catastrophizing and fear of impending death. Such excessive and disproportionate thoughts associated with somatic symptoms are one central feature of somatic symptom disorders. These disorders, frequently referred to as “functional somatic syndromes,” used to be negatively defined by “medically unexplained symptoms.” Compared with the older category of “somatoform disorders,” the construct of “somatic symptoms disorders” more accurately acknowledges that these symptoms may or may not be associated with organic lesions. The rarity of the ENS after inferior turbinate resection suggests that anatomical changes may not fully account for the symptoms and their functional
273.e10
C. Lemogne et al. / General Hospital Psychiatry 37 (2015) 273.e9–273.e10
impact [2]. Functional brain imaging rather suggests that ENS may share some psychophysiological mechanisms, including increased limbic reactivity, with other functional somatic syndromes such as fibromyalgia and irritable bowel syndrome [4,5]. Diagnosing the patient with a somatic symptom disorder was helpful in implementing efficient treatment, based on four principles: building and maintaining therapeutic alliance despite initial skepticism; targeting core dysfunctional beliefs (e.g., that ENS symptoms are predominantly caused by anatomical changes) with cognitive therapy; eradicating avoidance behaviors (i.e., those aiming at decreasing ENS symptoms) with behavioral therapy; prescribing a serotonin reuptake inhibitor, even in the absence of comorbid anxiety or depression. This strategy is efficient in treating fibromyalgia [6] or irritable bowel syndrome [7]. None of these interventions had been proposed to the patient until he was diagnosed with a somatic symptom disorder. Although therapeutic alliance is warranted in any patient–doctor relationship [8], it is especially challenging to achieve with patients with somatic symptom disorders who are typically reluctant to any psychological account of their symptoms. Because of the painful nature of the symptoms, a serotonin and norepinephrine reuptake inhibitor was preferred and introduced at low dosage (i.e., venlafaxine 37.5 mg). The patient progressively stepped back from dysfunctional beliefs through standard cognitive therapy techniques (e.g., socratic questioning) while becoming able to identify and leave several avoidance behaviors. These behaviors were nasal irrigations and the use of an air humidifier (aiming at reducing nasal dryness), repeated medical consultations (aiming at reducing the fear of having an undiagnosed hazardous physical condition) and physical inactivity (aiming at reducing dyspnea). Venlafaxine was gradually increased up to 150 mg over 3 months, with weight gain and a transient increase of nose dryness as side effects. Although the patient did not report much improvement of ENS symptoms per se, dramatic functional improvement was observed between month 2 and month 4 and confirmed by patients' relatives. At month 6, he was working again, displaying back-to-normal levels of functioning in social and leisure activities and was no longer seeking care for ENS. At month 12, venlafaxine was gradually stopped, without functional relapse at month 18.
Although many clinicians emphasize the potential role of psychological mechanisms in the ENS etiology and impact, targeting these mechanisms is rarely considered as central. Although we cannot determine the respective weight of cognitive behavior therapy and venlafaxine in the remediation of this patient, this case report suggests that treating ENS as a somatic symptom disorder might constitute a first-line, safe alternative to surgical or even medical treatment. It is noteworthy that eradicating avoidance behaviors may conflict with usual recommendations (e.g., nasal irrigations) but is critical as they may maintain ENS symptoms through operant conditioning. Future studies are warranted to establish the efficacy and safety of this approach.
Acknowledgments Conflicts of interest: None. Funding source: None.
References [1] Chhabra N, Houser SM. The diagnosis and management of empty nose syndrome. Otolaryngol Clin North Am 2009;42:311–30. [2] Sozansky J, Houser SM. Pathophysiology of empty nose syndrome. Laryngoscope 2015;125:70–4. [3] Jiang C, Wong F, Chen K, Shi R. Assessment of surgical results in patients with empty nose syndrome using the 25-item Sino-Nasal Outcome Test Evaluation. JAMA Otolaryngol Head Neck Surg 2014;140:453–8. [4] Browning M, Fletcher P, Sharpe M. Can neuroimaging help us to understand and classify somatoform disorders? A systematic and critical review. Psychosom Med 2011; 73:173–84. [5] Freund W, Wunderlich AP, Stöcker T, Schmitz BL, Scheithauer MO. Empty nose syndrome: limbic system activation observed by functional magnetic resonance imaging. Laryngoscope 2011;121:2019–25. [6] Clauw DJ. Fibromyalgia: a clinical review. JAMA 2014;311:1547–55. [7] Ford AC, Quigley EMM, Lacy BE, Lembo AJ, Saito YA, Schiller LR, et al. Effect of antidepressants and psychological therapies, including hypnotherapy, in irritable bowel syndrome: systematic review and meta-analysis. Am J Gastroenterol 2014;109: 1350–65. [8] Attale C, Lemogne C, Sola-Gazagnes A, Guedeney N, Slama G, Horvath A-O, et al. Therapeutic alliance and glycaemic control in type 1 diabetes: a pilot study. Diabetes Metab 2010;36:499–502.