International Journal of Pediatric Otorhinolaryngology 74 (2010) 1196–1198
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Case report
Haloperidol: A possible medication for the treatment of exacerbation of intractable psychogenic sneezing Sukru Nail Guner a,*, Cem Gokcen b, Bahar Gokturk a, Ozgul Topal c a
Department of Pediatrics, Baskent University Medical Faculty, Konya, Turkey Department of Child and Adolescent Psychiatry, Meram Education and Research Hospital, Konya, Turkey c Department of Otolaryngology, Baskent University Medical Faculty, Konya, Turkey b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 29 April 2010 Received in revised form 12 July 2010 Accepted 15 July 2010 Available online 11 August 2010
Sneezing is one of the physiological defense mechanisms that develops generally due to nasal irritation. But intractable sneezing episodes are uncommon and generally detected among the adolescents. It is difficult to distinguish physiologic sneezing from psychogenic sneezing. Herein, we report a 12-year-old girl who was complaint with intractable sneezing. She was diagnosed as intractable psychogenic sneezing and haloperidol treatment was started. All symptoms had resolved completely within 2 weeks. As a result, haloperidol can be considered as a different treatment modality for intractable psychogenic sneezing. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Haloperidol Intractable sneezing Psychogenic factors
1. Introduction Sneezing is a coordinated protective response to the irritation of the upper respiratory airways, especially the nasal cavity. The sneezing reflex frequently accompanies allergic and non-allergic rhinitis. It can also be triggered by odors, chemical irritants, infections, bright light or sunlight, physiologic stimulus applied to the trigeminal nerve, psychiatric or central nervous system pathologies, and sexual ideations [1–3]. ‘‘Psychogenic sneezing’’ was first described by Shilkret in 1949 in a 40-year-old woman [4], and approximately 50 cases were reported thereafter. The majority of the cases are adolescents [2,5,6]. Physiologic sneezing can be distinguished from psychogenic sneezing upon careful observation. Intractable psychogenic sneezing (IPS) differs from a real sneeze with the following features (Table 1): (1) a growl sound is heard intranasally after a short breathing phase in IPS; (2) IPS has a different rhythm and frequency; (3) while the eyes remain open in IPS, keeping the eyes open in real sneezing is difficult [7]; (4) IPS disappears during sleeping and eating and when the attention is removed [1,8]; (5) the sneezing in IPS is generally refractory to a wide variety of medications but responds well to psychological measures [6,8]; and (6) although there is a repetitive sneezing, no nasal congestion, nasal itching or rhinorrhea is seen, as would be observed in allergic rhinitis.
* Corresponding author at: Mimar Sinan Mh. 177, Sk. No: 1/15, 55200 Samsun, Turkey. Tel.: +90 362 3121919; fax: +90 362 4576041. E-mail address:
[email protected] (S.N. Guner). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.07.012
We reported an adolescent girl with intractable psychogenic sneezing who was treated with haloperidol. 2. Case description A 12-year-old girl was admitted to our clinic at May with the complaint of sneezing recurring consecutively 15–20 times per minute, which had started abruptly in the last 24 h. She had been admitted to emergency service with complaints of palpitation, shortness of breath, and feeling faint when the sneezing intensified. Despite attempted treatments with antihistaminic agents, oral/intranasal steroids, inhaled bronchodilators, and intranasal anesthesia, no response was obtained. There was no accompanying nasal discharge, itchy nose or eyes, cough, fever, headache, snoring, or constant clearing of the throat, and she had no history of frequent sore throats. Her medical history revealed that she had experienced intermittent sneezing that required antihistaminic drugs and intranasal steroid during the spring for 2 years. She had a history of stuttering since the age of 4 years, which had become aggravated recently. Two months before the current admission, 50 mg/day sertraline treatment had been started after she was diagnosed with anxiety following the clinical interview and completion of the State-Trait Anxiety Inventory (STAI) in a pediatric Psychiatry Department. On physical examination, she had normal growth, and her vital signs were within normal limits. She had intractable and recurrent paroxysms of sneezing, but we observed that she made a sound like a sneeze in a short and frequent manner with eyes opened and she looked around her during the examination. She had a mild
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Table 1 Differential diagnosis between physiologic sneezing and psychogenic sneezing. Physiologic sneezing
Intractable psychogenic sneezing (IPS)
In all ages and sexes Semi-autonomous, convulsive expulsion of air following a deep inspiration
Generally 10–14 years/female predominance Intranasal growl sound following a short breathing phase with an abnormal rhythm and frequency Minimal aerosolization of secretions Eyes remain open with minimal facial expression Sneezing disappears while the attention is removed (exp: sleeping, eating) No signs of physical examination Normal test results No respond to adequate conventional pharmacotherapy
Abundantly aerosolization of secretions It is difficult to sneeze with open eyes Sneezing continues while the attention is removed Pathologic sings of underlying disorders in physical examination Positive diagnostic test results Respond to the treatment of the underlying disorders
nasal mucosal pallor but no nasal congestion, nasal itching, rhinorrhea, postnasal drainage, cough, periorbital edema, or transverse crease at the nose secondary to the allergic salute. The rest of her physical examination was unremarkable. Complete blood count, serum IgE level, total eosinophil count, serum electrolytes, and liver and renal function tests were within normal limits. No abnormalities were determined during the rhinoscopic nasal examination, audiometric evaluation and respiratory function tests. Radioallergosorbent test (RAST) results of aeroallergens were all negative. Skin prick test was positive to D. pteronyssinus and cockroach. Paranasal sinus tomography was normal except for the presence of right nasal deviation and bullous appearance in the lamellar part of the right middle concha. Despite the history of allergic rhinitis during the last 2 years and positive skin prick test, we did not observe any positive physical examination findings except a mild nasal mucosal pallor. Moreover, there had been no response to the allergy treatment. After administering 5 mg diazepam intravenously, the sneezing was stopped during sleep and resumed a short time after awakening. It was observed that she did not sneeze when she was alone. When evaluated in the Psychiatry Department, it was ascertained that her symptoms had begun shortly after a stress that developed due to a decline in her school performance. No other tic behavior except for making sounds was detected during the interview with the patient based on the DSM IV (Diagnostic and Statistical Manual of Mental Disorders). Haloperidol (1 mg/day) was added to the sertraline treatment (50 mg/day) and the signs had noticeably reduced in 3–4 days. All symptoms had resolved completely within 2 weeks. Haloperidol was gradually decreased and stopped at the end of the first month, and only sertraline treatment was continued. No side effects of haloperidol were observed owing to low dose and short-term intake. The sertraline and the psychotherapeutic interview with the patient and the parents were carried out to prevent the recurrence the disease. The patient was free of signs and symptoms during 6-month follow-up. 3. Discussion Intractable psychogenic sneezing is a diagnosis of exclusion, so the patients must be evaluated carefully. Many factors in the patient’s history, such as allergy, infection, convulsion, vocal tics (Tourette syndrome), medications used, psychiatric problems, psychosocial development, role models related to the disease, and school performance were questioned [5]. Allergic rhinitis is one of the most important causes of sneezing [9]. However, to the best of our knowledge, there has been no previous report showing allergic rhinitis as a cause of intractable and paroxysmal sneezing. Our case had been treated previously for an allergic rhinitis with a mild clinical course. Recently, there had been no difference in the environmental conditions that would explain an increase in her complaints. In addition, in the nasal examination of the patient, there was no nasal discharge, mucosal hyperemia or conchal hypertrophy. Intranasal anesthesia was
applied with 10% lidocaine for exclusion of the mechanical and irritative causes of sneezing, but all the symptoms persisted. The frequency and severity of our patient’s sneezing was irregular. She made a sound similar to a sneeze sometimes 20–30 times consecutively and other times only 8–10 times consecutively per minute. In a 24-h observation, it was noticed that she did not sneeze during her sleep and started to sneeze just a few hours after awakening. It was also observed that she followed the events occurring in her surroundings with her eyes open while sneezing and that she did not sneeze when she was alone. Furthermore, the patient showed no apparent discomfort during the examinations and appeared to welcome the attention. Different treatment modalities, such as intranasal cocainebenzocaine, oral chlorpromazine and phenytoin have been used to stop sneezing attacks [2]. However, an underlying cause having a psychological rather than a somatic basis would give rise to treatment failure. Shapiro [10] determined that the symptoms disappeared after intranasal anesthesia application in two cases who had sneezing during sleep, and they emphasized that this could help in the differential diagnosis of psychogenic-paroxysmal sneezing. In our case, different treatment modalities in different medical centers were applied but no acceptable results were obtained. In our clinics, intranasal lidocaine (Xylocaine1 10% spray) was also applied, but there was no change in the sneezing frequency. It is known that stuttering and anxiety are closely related [11]. The selective serotonin reuptake inhibitors are being used frequently in anxiety disorders in addition to stuttering and speech disorders, and they are effective by decreasing the present anxiety [12]. Paroxysmal sneezing is similar to the other forms of repeating central nerve system discharge such as tics and other types of involuntary movements. Haloperidol is a sedative, antipsychotic drug that can be used in conditions such as stuttering and intractable hiccup. Davison [13] reported a resolution of symptoms using haloperidol treatment in increasing doses in a 60-year-old woman who admitted with a complaint of intractable sneezing, but after drug cessation, the complaint resumed. As haloperidol is a potent dopamine-receptor blocker, paroxysmal sneezing could be based on a disturbance in the cerebral dopaminergic transmission system [13]. However, in our presented case, obvious improvement shortly after starting haloperidol treatment and no recurrence of the symptoms following the drug removal led us to think that the sedative effect of haloperidol proved useful in this situation rather than its anti-dopaminergic effect. 4. Conclusion Intractable paroxysmal sneezing is a disorder observed especially in adolescents, and failure of medical treatment can lead to severe anxiety or fear for the parents and the family. The cases with IPS are generally diagnosed after a long follow-up period. This case is important because an accurate diagnosis was established in a short period and the condition could be treated
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successfully with haloperidol. Although it is not exactly clear via which mechanism haloperidol contributed to the treatment, it should be considered among the treatment choices especially for the exacerbation of IPS. Determination of the underlying factors triggering this psychogenic-based disorder with a detailed anamnesis can prevent unnecessary and expensive studies to reach the diagnosis. References [1] T.J. Lin, C.A. Maccia, C.G. Turnier, Psychogenic intractable sneezing: case reports and a review of treatment options, Ann. Allergy Asthma Immunol. 91 (2003) 575– 578. [2] L.J. Fochtmann, Intractable sneezing as a conversion symptom, Psychosomatics 36 (March) (1995) 103–112. [3] B. Niggemann, Functional symptom confused with allergic disorders in children and adolescent, Pediatr. Allergy Immunol. 13 (2002) 312–318.
[4] H.H. Shilkrel, Psychogenic sneezing and yawning, Psychosom. Med. 11 (1949) 127–128. [5] P. Gopalan, S.T. Browning, Intractable paroxysmal sneezing, J. Laryngol. Otol. 116 (2002) 958–959. [6] D. Wiener, K. McGrath, R. Patterson, Factitious sneezing, J. Allergy Clin. Immunol. 75 (1985) 741–742. [7] G.E. Bergman, L.B. Hiner, Psychogenic intractable sneezing in children, J. Pediatr. 105 (1984) 496–498. [8] M.U. Keating, E.J. O’Connell, M.I. Sachs, Intractable paroxysmal sneezing in an adolescent, Ann. Allergy 62 (1989) 429–431. [9] O. Pfaar, U. Raap, M. Holz, K. Ho¨rmann, L. Klimek, Pathophysiology of itching and sneezing in allergic rhinitis, Swiss Med. Wkly 139 (2009) 35–40. [10] R.S. Shapiro, Paroxysmal sneezing in children: two new cases, J. Otolaryngol. 21 (1992) 437–438. [11] K. Mulcahy, N. Hennessey, J. Beilby, M. Byrnes, Social anxiety and the severity and typography of stuttering in adolescents, J. Fluency Disord. 33 (2008) 306–319. [12] E.G. Conture, R.F. Curlee, Stuttering and Related Disorders of Fluency, third edition, Thieme, 2007. [13] K. Davison, Pharmacological treatment for intractable sneezing, Br. Med. J. (Clin. Res. Ed.) 284 (1982) 1163–1164.