Spasmodic dysphonia

Abstract

Stridor as an initial symptom of a conversion reaction (hysteria) is rare. We report cases of hysterical stridor in two older women, unrelated and unacquainted, from the same rural community in Oman. Once the diagnosis was made, both patients were successfully treated with a single dose of an anxiolytic. We also review the literature on hysterical stridor and discuss the diagnostic dilemmas and therapeutic options.

Introduction

Somatoform disorders are characterized by physical symptoms for which no adequate medical explanation can be found. Symptoms and complaints are serious enough to cause significant emotional distress or impair a patient's ability to function in social and occupational roles. These disorders have been grouped under the term hysteria (Gr. hystera womb), as they were once incorrectly thought to affect only women.

One subtype of somatoform disorder is called conversion disorders. They are characterized by one or more unexplained neurologic complaints, such as paralysis, blindness, and mutism. By definition, conscious malingering and factitious disorders are excluded. (1) In this article, we describe two cases of conversion disorder in older women that manifested as stridor. We also discuss the diagnostic dilemma posed by this essentially benign condition, and we review the treatment options.

Case reports

Patient 1. A 62-year-old woman from a rural community was escorted to the Ibri Regional Referral Hospital in Oman by her primary care physician on June 12, 2002, for evaluation of a 1-day history of stridor. In view of the severity of her stridor and the presence of signs of respiratory distress (i.e., tachypnea and flaring of the alae nasi), she was transferred to the emergency department for intensive monitoring.

An awake fiberoptic laryngoscopy revealed that the patient had a normal larynx but paradoxical vocal fold movement during inspiration. Findings on radiologic investigations and blood gas parameters were within normal limits. Nevertheless, empiric treatment with an injectable steroid and nebulized epinephrine under monitoring was administered.

When these medications failed to relieve the stridor, preparations were made for surgical intervention. However, at that point, the patient's son remarked that his mother had experienced a similar episode of stridor 2 weeks earlier when she had gone to the hospital to visit her sister, who had been injured in an automobile accident. That episode of stridor had subsided spontaneously. The son then reported that the current episode had been precipitated when his mother had been informed that the same sister had been involved in another accident as she returned home after discharge. At that point, we considered a diagnosis of hysterical stridor.

As a trial, we gave the patient a 5-mg dose of the anxiolytic midazolam under careful monitoring. This resulted in almost instantaneous relief of her stridor. The patient was discharged after an overnight stay and a consultation with a psychiatrist. No comorbid psychiatric condition was identified.

Patient 2. A 53-year-old woman from the same rural community came to the ENT outpatient department on June 19, 2002, with a complaint of aphonia of 2 days' duration. Our attempt to examine her vocal folds by indirect laryngoscopy provoked an attack of severe and unrelenting stridor.

The patient was transferred to the emergency department for monitoring. An awake fiberoptic laryngoscopic examination revealed an essentially normal larynx and mildly paradoxical vocal fold movement. Having seen a case of hysterical stridor just 1 week earlier, our index of suspicion was high. Again, 5 mg of midazolam was administered intravenously under careful monitoring. The patient's symptoms resolved immediately.

The patient was kept overnight, and a psychiatric evaluation the next day revealed that she had a major depressive illness, consequent to the death of her husband. She was discharged on an antidepressant and is being followed up by her primary care physician because a recurrence of her symptoms is deemed likely.

Discussion

Stridor is a sign of upper airway obstruction, and it merits immediate attention on the part of the emergency resuscitation team. While attempts should be made to diagnose the cause of stridor, concurrent management should be aggressive, given the life-threatening nature of this condition. Medical measures (e.g., a corticosteroid and nebulization with racemic epinephrine) and/or surgical measures (e.g., intubation, cricothyrotomy, and tracheostomy) should be part of the emergency department treatment protocol. (2)

Anecdotal reports and reviews in the pediatric, (3-8) pub monary, (9-15) psychiatric, (16,17) and otolarynologic (18-26)literature describe a psychosomatic illness that manifests as spontaneously resolving stridor. This is a conversion type of somatoform disorder, for which a variety of synonyms has been used in the literature (table). (2) In this disorder, awake fiberoptic examination will reveal a normal larynx and paradoxical vocal fold movement--that is, adduction during inspiration or excessive adduction of the vocal folds during expiration. (27) Consequently, the patient might experience inspiratory stridor, expiratory stridor, or an overlapping stridor with features of both. (2) Flow-volume loops will reveal a pattern of variable extrathoracic obstruction with diminished peak inspiratory flow. (27)

Although the etiology is most often functional, organic causes should be ruled out. A classification scheme proposed by Maschka et al attempts to incorporate all causes, both organic and functional: (1) brainstem compression, (2) cortical or upper motor neuron injury, (3) nuclear or lower motor neuron injury, (4) vocal fold movement disorders, (5) gastroesophageal reflux, (6) factitious or malingering disorders, and (7) somatization/conversion disorder. (28) It has been shown that sedation in the emergency department and general anesthesia in the operating room cause the vocal folds to relax in patients with functional stridor, but it has no such effect in patients with organic vocal fold paralysis; therefore, sedation or anesthesia can be used to differentiate the two entities. (2)

Clinically, the diagnosis of conversion disorder is one of exclusion. (29) Misdiagnoses and delays in diagnosis have led to unnecessary tracheostomies and intubations in many cases. (17) A high index of suspicion for conversion disorder manifesting as stridor should reduce unnecessary interventions. (27)

The two patients described in this article came to us within 1 week of each other. Although they lived in the same rural community, they were unrelated and unacquainted with each other. Awake fiberoptic intubation in both cases revealed paradoxical vocal fold movements. The severity of symptoms in the first patient resulted in the administration of an injectable steroid, and she was on the verge of undergoing a tracheostomy when her son reported her previous episode of stridor. With regard to the second patient, a higher index of suspicion enabled us to avoid unnecessary medication. Both patients responded well to anxiolytic therapy. Only one of them (patient 2) had a comorbid condition (depression) and lacked an identifiable precipitating factor. Hence, she is undergoing close follow-up by her primary care physician.

The fourth edition of the American Psychiatric Association's Diagnostic and Statistical Manual of Mental Disorders (DSM-IV) lists five specific somatoform disorders: (1) somatization disorders, which are characterized by many physical complaints that affect many organ systems, (2) hypochondrias, which are characterized by a belief by the patient that some specific disease exists, (3) body dysmorphic disorder, which is characterized by a false or exaggerated belief that some body part is defective, (4) pain disorder, which is characterized by pain that is solely related to or significantly exaggerated by psychological factors, and (5) conversion disorder, which is characterized by one or more neurologic complaints (e.g., paralysis, blindness, and paresthesias) that cannot be explained by any known neurologic disorder. (30) In addition, the diagnosis of a conversion disorder requires that psychological factors are associated with the initiation or exacerbation of symptoms. These disorders occur more often in wo men than in men and more often in adolescents and young adults than in older adults. Conversion disorders are also frequently associated with comorbid conditions such as depression, anxiety, or schizophrenia. (1) Initial airway symptoms include aphonia, dysphonia, dyspnea with or without stridor, apnea, and apparent unconsciousness. (27) The acute manifestations of conversion disorders usually resolve spontaneously within a few days, although some cases take weeks to resolve. Resolution is facilitated by insight-oriented supportive or behavioral therapy in the setting of an authoritative therapeutic relationship. Symptoms can recur during periods of stress. A good prognosis is associated with the presence of an identifiable stressor, a sudden onset of symptoms, a good premorbid adjustment, and a lack of comorbid psychiatric or medical disorders. (1)

Acknowledgments

The authors thank the director of General Health Services, the superintendent of Planning and Training for the Al Dhahira Region, and the medical superintendent of the Ibri Regional Referral Hospital for permitting and facilitating this report.

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From the Department of Otolaryngology (Dr. Nayar and Dr. Zanak) and the Department of Psychiatry (Dr. Ahmed), Ibri Regional Referral Hospital, Al Dhahira Region, Sultanate of Oman.

Reprint requests: Dr. Ravi C. Nayar, Head, Department of Otolaryngology, Ibri Regional Referral Hospital, P.O. Box 46, Postal Code 516, Al Dhahira Region, Sultanate of Oman. Phone: 968-650-855, ext. 222; fax: 968-489-725; e-mail: ravi23@omantel.net.om

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