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Psychogenic intractable sneezing in children
BEHAVIORAL PEDIATRICS
Psychogenic intractable sneezing in children Four preadolescent children had paroxysms o f intractable sneezing. After days o f hospitalization and numerous local and systemic drug treatments had failed, a psychogenic cause of their problem was entertained. This uncommon form o f conversion reaction most often occurs between the ages o f 9 and 15 years, and prognosis is good i f the appropriate interventions are undertaken. (J PeDIATR 105:496, 1984)
Garrett E. Bergman, M.D., and Linda B. Hiner, M.D. PROLONGED INTRACTABLE SNEEZING is unusual. P r e vious r e p o r t s h a v e d e m o n s t r a t e d t h e incidence o f this conversion r e a c t i o n to be h i g h e s t in y o u n g adolescents. W e have seen four y o u n g c h i l d r e n in w h o m i n t r a c t a b l e sneezi n g was initially m i s m a n a g e d with h o s p i t a l i z a t i o n a n d n u m e r o u s i n t e r v e n t i o n s d i r e c t e d at t h e s y m p t o m r a t h e r t h a n at its cause. By r e c o g n i z i n g t h e p s y c h o g e n i c origin o f this p r o b l e m , t h e p e d i a t r i c i a n can quickly institute m o r e effective b e h a v i o r a l intervention. CASE REPORTS
Patient 1. This I I-year-old girl was admitted to the hospital after 3 days of sneezing every few seconds when awake. She did not stop sneezing after local application of sympathomimetic agents or corticosteroids, systemic administration of antihistamines, or sedation. Exposure to an insecticide seemed to precipitate the attack. Past medical history included sinusitis, migraine headache, and resolved enuresis and encopresis. Her father and twin brother both had an allergic diathesis. The child's sneezing consisted of a repetitive, head-bobbing snort not preceded by a large intake of breath. The alae nasae were erythematous. No nasal foreign body or swollen turbinates were found. During her hospitalization she sneezed more frequently when she was upset or embarrassed. On the sixth day of continuous sneezing, her previously aloof mother accepted the emotional basis of the problem. As plans for outpatient psychotherapy were made, the sneezing stopped abruptly and did not recur. Patient 2. This 12-year-old girt was admitted to the hospital after 2 days of sneezing not responsive to injected epinephrine, intranasal sympathomimetics, or parenteral sedation (barbiturate, From the Department o f Pediatrics, The Medical College o f Pennsylvania. Submitted for publication Jan. 23, 1984; accepted Feb. 24, 1984. Reprint requests: Garrett E. Bergman, M.D., Department o f Pediatrics, The Medical College o f Pennsylvania, 3300 Henry Ave., Philadelphia, PA 19129.
496
TheJournalofPEDlATRICS
Philadelphia, Pennsylvania
phenothiazine). Occasional sneezing had been precipitated recently by the odor of furniture cleaner. One day prior to admission she was sent home by the school nurse for intractable sneezing. The child had been healthy, with no recent respiratory infections and no known allergies. The child's mother had asthma, and one sibling had allergic rhinitis. Her family was unstable: her mother had been married three times; her old.eat brother had recently entered a correctional facility; and she did not get along well with her other siblings. On admission the child sneezed three to eight times every 15 to 30 seconds. Her nares were inflamed and the nasal mucosa slightly hyperemic. There was no intranasal foreign body. Her quiet, abbreviated sneezes often sounded like nasal coughing; no nasal secretions were formed. After admission she stopped sneezing immediately after her mother left, and shortly thereafter she slept. The next day she was withdrawn but sneezed regularly when observed by hospital staff or visited by family members. Her sneezing stopped completely after 2 days, recurring only when she was anxious. After several weeks of outpatient psychotherapy, sneezing paroxysms could be precipitated by asking about the symptoms. Patient 3. This 9-year-old boy was admitted to the hospital after 10 hours of intractable sneezing, which had evolved from brief paroxysms every 5 minutes to longer paroxysms every 1 to 2 minutes. Symptoms did not improve after nasal instillation of neosynephrine, cocaine, or dexamethasone; after inhalation of cool mist; or after systemic administration of an antihistamine and a sedative. The patient bad asthma and allergic rhinitis, and had been enuretic until 81/2 years of age. Physical examination revealed no sign of a respiratory infection, no nasal foreign body, and no wheezing. His parer~ts were divorced. The child had been living with his father until 2 weeks prior to this admission, when he and three siblings had moved to their mother's home. During his hospital stay the frequency of his sneezing increased whenever he was aware of being observed; he sneezed only infrequently when left alone. His mother accepted the emotignal basis of his sneezing and was amenable to therapy. When the patient became aware of his approaching discharge, he developed continuous coughing, and the sneezing stopped. The cough was very exaggerated when physicians, nurses, or his mother attended him, and was very
Volume 105 Number 3
infrequent when he was isolated by closing the door to his room. The coughing stopped in 1 day, and he was discharged for outpatient medical care and psychotherapy. The sneezing never recurred. Patient 4. This 10-year-old boy was admitted after 4 days of spasmodic sneezing not responsive to intranasally administered sympathomimetic drugs or corticosteroids, to injected long-acting epinephrine, or to systemically administered antihistamines or sedatives (diazepam). Sneezing occurred in bursts of 6 to 10 abortive nasal "'snorts," initially at long intervals, but gradually becoming more frequent (every 5 minutes) and intense. Various physical stimuli could precipitate a paroxysm. Sneezing ceased during sleep but increased when he was being attended by his mother. On the day of admission he had had a paroxysm of more than 200 consecutive sneezes (counted by his mother). The patient was an only child, in good general health. He had no known allergies, but an aunt had hay fever and his father had recurrent sinusitis. Earlier that year the patient had been in psychotherapy for school refusal, and was receiving medication on occasion for "irritable stomach." Physical examination revealed normal findings except for mild hyperemia of the nasal turbinates. No nasal foreign body was present. After admission the sneezing spells became infrequent and stopped. His mother was very apprehensive during his hospitalization and regarding his discharge. He was discharged with plans to reinstitute psychotherapy. Sneezing did not recur. DISCUSSION The physiologic reflex mechanism of sneezing has been reviewed by Brubaker, ~ Kofman, 2 and Co? Stimuli that can induce sneezing include local nasal stimulation (odors, irritants, allergies, infections,4 tumors, foreign bodies), visual stimulation (bright light), vasomotor nasal congestion (chilling or drying of inhaled air, emotional or sexual excitation, postprandial satiation), pregnancy, menstruation, and temporal lobe epilepsy. Intractable paroxysms of sneezing are unrelated to these usual causes, although many patients have had allergic diathesis; in one patient, IgE-mediated sensitivity to trieth~nolamine was implicated. 5 A normal physiologic sneeze (short rapid involuntary inspiration followed immediately ~ forceful involuntary expiration through the nasopharyn:~) may not be seen in intractable repetitive paroxysms of sneezing; rather, there may be an "aborted" or "pseudosneeze ''6 (little inspiratory phase, short nasal grunting sound, and little or no aerosolization of nasal mucous secretions). These "sneezes" are usually repeated 3 to 30 times per minute, for hours or days, and may be relieved only by sleep. Social isolation of the patient may diminish the frequency of the sneezes. Similar to a tic disorder, this clinical pattern can resemble but be distinguished from Tourette syndrome 7 by the lack of associated findings and course. The clinical findings in our four patients and reports of
Psychogenic intractable sneezing
497
other patients lead us to conclude that this is a typical, albeit uncommonly recognized, conversion reaction. Maloney 8 has reviewed the definition of conversion reactions in children, stressing the characteristic "disabilities in various sensory, visceral or voluntary motor systems of the body; with unconscious symbolic meaning; [but with] no organic pathology." Children he observed with this form of psychosomatic illness had often suffered a recent family crisis, were inhibited from discussing the emotional stress they felt, and were likely harboring unresolved grief reactions. In a recent review of 20 patients with intractable sneezing, 15 were teenagers aged 17 years or younger. The preponderance of young adolescents, aged 9 to 15 years, among identified patients 2.3"6-~ has not been noted in the pediatric literature. Numerous attempts at treatment with topical agents (antihistamines, steroids, cocaine, sympathomimetics) and systemic drugs (barbiturates, phenothiazines, antihistamines, benzodiazepines) have been unsuccessful in reported patients and in our patients. The possibility of a psychogenic origin is usually considered only after an unsuccessful series of attempts to treat paroxysmal sneezing as a physical disease. Secondary gain is not considered an attribute of conversion symptomsY 2 Our experience and that of others ~2'~3 leads us to believe that it is not helpful to focus on the dramatic symptom. Meeting with the family and child to explore, in a delicate manner, unrecognized or unacknowledged stresses often allows resolution of the subconsciously generated symptom. 7 Even weeks after acute sneezing paroxysms had resolved, we inadvertently reinduced the symptom in one patient by asking about her sneezing. This should not be surprising, because of the extreme suggestibility of patients with conversion reaction. The prognosis for intractable sneezing is very good. Our patients and those previously reported all recovered within days to months. Occasional patients have received widespread publicity in the lay press, which has provided a large degree of secondary gain and unnecessary prolongation of symptoms. Hypnosis, drugs, aversive conditioning, family therapy, and individual psychotherapy have all been used successfully, but eliminating the symptom is only part of the necessary treatment. In some cases, meeting with the family two or three times to explore areas of stress may suffice to reduce the family discord that precipitated the symptomS; in other cases, psychiatric intervention will be needed. In no case report was recurrence a problem, nor was it in our patients. Long-term foll0w-up of 34 children and adolescents with hyperventilafion syndrome, another type of conversion reaction, demonstrated a high incidence of
498
Bergman and Hiner
chronic severe anxiety and persistence of hyperventilation. ~4 Thus, the prognoses in these two entities, similar in many respects, would appear to differ, By recognizing this clinical picture of intractable, paroxysmal sneezing in a young adolescent as a conversion reaction, the pediatrician can appropriately focus therapeutic interventions at the underlying emotional basis of the problem. Outpatient treatment and psychotherapies of various types have been successful; in all reported cases symptoms abate in a short time if secondary gain is eliminated. Unnecessary and potentially harmful hospitalization, diagnostic testing, and treatment with physical agents can then be avoided. We thank Victor C. Vaughn, M.D., Vera Mallsoff, M.D., and Estelle Gold, M.D., for permission to report on their patients. REFERENCES 1. Brubaker A: The physiology of sneezing. JAMA 73:585, 1919. 2. Kofman O: Paroxysmal sneezing. Can Med Assoc J 91:154, 1964. 3.~"Co S: Intractable sneezing: Case report and literature review. Arch Neurol 36:111, 1979.
The Journal of Pediatrics September 1984 4. Rambar AC: Paroxysmal sneezing in whooping cough J PEDIATR 8:582, 1936. 5. Herman J J: Intractable sneezing due to IgE-mediated triethanolamine sensitivity. J Allergy Clin Immunol 71:339, 1983. 6. Elkins M, Milstein J J: Hypnotherapy of pseudo-sneezing: A case report. Am J Clin Hypn 4:273, 1962, 7. Vogel DH: Otolaryngologic presentation of tic-like disorders. Laryngoscope 89:1474, 1979. 8. Maloney M J: Diagnosing hysterical conversion reactions in children. J PEDIATR97:1016, 1980. 9. Gallia L J, Roscoe G: Intractable sneezing. Trans Pa Acad Ophthalmol Otolaryngol 34:164, 1981. 10. Murray N, Bierer J: Prolonged sneezing: A case report. Psychosom Med 13:56, 1951. 1 l. Kaplan M J, Lanoff G: Intractable paroxysmal sneezing. A clinical entity defined with case reports. Ann Allergy 28:24, 1970. 12. Lazare A: Current concepts in psychiatry: Conversion symptoms. N Engl J Med 305:745, 1981. 13. Murphy GE: The clinical management of hysteria. JAMA 247:2559, 1982. 14. Herman SP, Stickler, GB and Lucas, AR: Hyperventilation syndrome in children and adolescents: Long term follow-up. Pediatr 67:183-187, 1981.
Psychogenic intractable sneezing in children Four preadolescent children had paroxysms o f intractable sneezing. After days o f hospitalization and numerous local and systemic drug treatments had failed, a psychogenic cause of their problem was entertained. This uncommon form o f conversion reaction most often occurs between the ages o f 9 and 15 years, and prognosis is good i f the appropriate interventions are undertaken. (J PeDIATR 105:496, 1984)
Garrett E. Bergman, M.D., and Linda B. Hiner, M.D. PROLONGED INTRACTABLE SNEEZING is unusual. P r e vious r e p o r t s h a v e d e m o n s t r a t e d t h e incidence o f this conversion r e a c t i o n to be h i g h e s t in y o u n g adolescents. W e have seen four y o u n g c h i l d r e n in w h o m i n t r a c t a b l e sneezi n g was initially m i s m a n a g e d with h o s p i t a l i z a t i o n a n d n u m e r o u s i n t e r v e n t i o n s d i r e c t e d at t h e s y m p t o m r a t h e r t h a n at its cause. By r e c o g n i z i n g t h e p s y c h o g e n i c origin o f this p r o b l e m , t h e p e d i a t r i c i a n can quickly institute m o r e effective b e h a v i o r a l intervention. CASE REPORTS
Patient 1. This I I-year-old girl was admitted to the hospital after 3 days of sneezing every few seconds when awake. She did not stop sneezing after local application of sympathomimetic agents or corticosteroids, systemic administration of antihistamines, or sedation. Exposure to an insecticide seemed to precipitate the attack. Past medical history included sinusitis, migraine headache, and resolved enuresis and encopresis. Her father and twin brother both had an allergic diathesis. The child's sneezing consisted of a repetitive, head-bobbing snort not preceded by a large intake of breath. The alae nasae were erythematous. No nasal foreign body or swollen turbinates were found. During her hospitalization she sneezed more frequently when she was upset or embarrassed. On the sixth day of continuous sneezing, her previously aloof mother accepted the emotional basis of the problem. As plans for outpatient psychotherapy were made, the sneezing stopped abruptly and did not recur. Patient 2. This 12-year-old girt was admitted to the hospital after 2 days of sneezing not responsive to injected epinephrine, intranasal sympathomimetics, or parenteral sedation (barbiturate, From the Department o f Pediatrics, The Medical College o f Pennsylvania. Submitted for publication Jan. 23, 1984; accepted Feb. 24, 1984. Reprint requests: Garrett E. Bergman, M.D., Department o f Pediatrics, The Medical College o f Pennsylvania, 3300 Henry Ave., Philadelphia, PA 19129.
496
TheJournalofPEDlATRICS
Philadelphia, Pennsylvania
phenothiazine). Occasional sneezing had been precipitated recently by the odor of furniture cleaner. One day prior to admission she was sent home by the school nurse for intractable sneezing. The child had been healthy, with no recent respiratory infections and no known allergies. The child's mother had asthma, and one sibling had allergic rhinitis. Her family was unstable: her mother had been married three times; her old.eat brother had recently entered a correctional facility; and she did not get along well with her other siblings. On admission the child sneezed three to eight times every 15 to 30 seconds. Her nares were inflamed and the nasal mucosa slightly hyperemic. There was no intranasal foreign body. Her quiet, abbreviated sneezes often sounded like nasal coughing; no nasal secretions were formed. After admission she stopped sneezing immediately after her mother left, and shortly thereafter she slept. The next day she was withdrawn but sneezed regularly when observed by hospital staff or visited by family members. Her sneezing stopped completely after 2 days, recurring only when she was anxious. After several weeks of outpatient psychotherapy, sneezing paroxysms could be precipitated by asking about the symptoms. Patient 3. This 9-year-old boy was admitted to the hospital after 10 hours of intractable sneezing, which had evolved from brief paroxysms every 5 minutes to longer paroxysms every 1 to 2 minutes. Symptoms did not improve after nasal instillation of neosynephrine, cocaine, or dexamethasone; after inhalation of cool mist; or after systemic administration of an antihistamine and a sedative. The patient bad asthma and allergic rhinitis, and had been enuretic until 81/2 years of age. Physical examination revealed no sign of a respiratory infection, no nasal foreign body, and no wheezing. His parer~ts were divorced. The child had been living with his father until 2 weeks prior to this admission, when he and three siblings had moved to their mother's home. During his hospital stay the frequency of his sneezing increased whenever he was aware of being observed; he sneezed only infrequently when left alone. His mother accepted the emotignal basis of his sneezing and was amenable to therapy. When the patient became aware of his approaching discharge, he developed continuous coughing, and the sneezing stopped. The cough was very exaggerated when physicians, nurses, or his mother attended him, and was very
Volume 105 Number 3
infrequent when he was isolated by closing the door to his room. The coughing stopped in 1 day, and he was discharged for outpatient medical care and psychotherapy. The sneezing never recurred. Patient 4. This 10-year-old boy was admitted after 4 days of spasmodic sneezing not responsive to intranasally administered sympathomimetic drugs or corticosteroids, to injected long-acting epinephrine, or to systemically administered antihistamines or sedatives (diazepam). Sneezing occurred in bursts of 6 to 10 abortive nasal "'snorts," initially at long intervals, but gradually becoming more frequent (every 5 minutes) and intense. Various physical stimuli could precipitate a paroxysm. Sneezing ceased during sleep but increased when he was being attended by his mother. On the day of admission he had had a paroxysm of more than 200 consecutive sneezes (counted by his mother). The patient was an only child, in good general health. He had no known allergies, but an aunt had hay fever and his father had recurrent sinusitis. Earlier that year the patient had been in psychotherapy for school refusal, and was receiving medication on occasion for "irritable stomach." Physical examination revealed normal findings except for mild hyperemia of the nasal turbinates. No nasal foreign body was present. After admission the sneezing spells became infrequent and stopped. His mother was very apprehensive during his hospitalization and regarding his discharge. He was discharged with plans to reinstitute psychotherapy. Sneezing did not recur. DISCUSSION The physiologic reflex mechanism of sneezing has been reviewed by Brubaker, ~ Kofman, 2 and Co? Stimuli that can induce sneezing include local nasal stimulation (odors, irritants, allergies, infections,4 tumors, foreign bodies), visual stimulation (bright light), vasomotor nasal congestion (chilling or drying of inhaled air, emotional or sexual excitation, postprandial satiation), pregnancy, menstruation, and temporal lobe epilepsy. Intractable paroxysms of sneezing are unrelated to these usual causes, although many patients have had allergic diathesis; in one patient, IgE-mediated sensitivity to trieth~nolamine was implicated. 5 A normal physiologic sneeze (short rapid involuntary inspiration followed immediately ~ forceful involuntary expiration through the nasopharyn:~) may not be seen in intractable repetitive paroxysms of sneezing; rather, there may be an "aborted" or "pseudosneeze ''6 (little inspiratory phase, short nasal grunting sound, and little or no aerosolization of nasal mucous secretions). These "sneezes" are usually repeated 3 to 30 times per minute, for hours or days, and may be relieved only by sleep. Social isolation of the patient may diminish the frequency of the sneezes. Similar to a tic disorder, this clinical pattern can resemble but be distinguished from Tourette syndrome 7 by the lack of associated findings and course. The clinical findings in our four patients and reports of
Psychogenic intractable sneezing
497
other patients lead us to conclude that this is a typical, albeit uncommonly recognized, conversion reaction. Maloney 8 has reviewed the definition of conversion reactions in children, stressing the characteristic "disabilities in various sensory, visceral or voluntary motor systems of the body; with unconscious symbolic meaning; [but with] no organic pathology." Children he observed with this form of psychosomatic illness had often suffered a recent family crisis, were inhibited from discussing the emotional stress they felt, and were likely harboring unresolved grief reactions. In a recent review of 20 patients with intractable sneezing, 15 were teenagers aged 17 years or younger. The preponderance of young adolescents, aged 9 to 15 years, among identified patients 2.3"6-~ has not been noted in the pediatric literature. Numerous attempts at treatment with topical agents (antihistamines, steroids, cocaine, sympathomimetics) and systemic drugs (barbiturates, phenothiazines, antihistamines, benzodiazepines) have been unsuccessful in reported patients and in our patients. The possibility of a psychogenic origin is usually considered only after an unsuccessful series of attempts to treat paroxysmal sneezing as a physical disease. Secondary gain is not considered an attribute of conversion symptomsY 2 Our experience and that of others ~2'~3 leads us to believe that it is not helpful to focus on the dramatic symptom. Meeting with the family and child to explore, in a delicate manner, unrecognized or unacknowledged stresses often allows resolution of the subconsciously generated symptom. 7 Even weeks after acute sneezing paroxysms had resolved, we inadvertently reinduced the symptom in one patient by asking about her sneezing. This should not be surprising, because of the extreme suggestibility of patients with conversion reaction. The prognosis for intractable sneezing is very good. Our patients and those previously reported all recovered within days to months. Occasional patients have received widespread publicity in the lay press, which has provided a large degree of secondary gain and unnecessary prolongation of symptoms. Hypnosis, drugs, aversive conditioning, family therapy, and individual psychotherapy have all been used successfully, but eliminating the symptom is only part of the necessary treatment. In some cases, meeting with the family two or three times to explore areas of stress may suffice to reduce the family discord that precipitated the symptomS; in other cases, psychiatric intervention will be needed. In no case report was recurrence a problem, nor was it in our patients. Long-term foll0w-up of 34 children and adolescents with hyperventilafion syndrome, another type of conversion reaction, demonstrated a high incidence of
498
Bergman and Hiner
chronic severe anxiety and persistence of hyperventilation. ~4 Thus, the prognoses in these two entities, similar in many respects, would appear to differ, By recognizing this clinical picture of intractable, paroxysmal sneezing in a young adolescent as a conversion reaction, the pediatrician can appropriately focus therapeutic interventions at the underlying emotional basis of the problem. Outpatient treatment and psychotherapies of various types have been successful; in all reported cases symptoms abate in a short time if secondary gain is eliminated. Unnecessary and potentially harmful hospitalization, diagnostic testing, and treatment with physical agents can then be avoided. We thank Victor C. Vaughn, M.D., Vera Mallsoff, M.D., and Estelle Gold, M.D., for permission to report on their patients. REFERENCES 1. Brubaker A: The physiology of sneezing. JAMA 73:585, 1919. 2. Kofman O: Paroxysmal sneezing. Can Med Assoc J 91:154, 1964. 3.~"Co S: Intractable sneezing: Case report and literature review. Arch Neurol 36:111, 1979.
The Journal of Pediatrics September 1984 4. Rambar AC: Paroxysmal sneezing in whooping cough J PEDIATR 8:582, 1936. 5. Herman J J: Intractable sneezing due to IgE-mediated triethanolamine sensitivity. J Allergy Clin Immunol 71:339, 1983. 6. Elkins M, Milstein J J: Hypnotherapy of pseudo-sneezing: A case report. Am J Clin Hypn 4:273, 1962, 7. Vogel DH: Otolaryngologic presentation of tic-like disorders. Laryngoscope 89:1474, 1979. 8. Maloney M J: Diagnosing hysterical conversion reactions in children. J PEDIATR97:1016, 1980. 9. Gallia L J, Roscoe G: Intractable sneezing. Trans Pa Acad Ophthalmol Otolaryngol 34:164, 1981. 10. Murray N, Bierer J: Prolonged sneezing: A case report. Psychosom Med 13:56, 1951. 1 l. Kaplan M J, Lanoff G: Intractable paroxysmal sneezing. A clinical entity defined with case reports. Ann Allergy 28:24, 1970. 12. Lazare A: Current concepts in psychiatry: Conversion symptoms. N Engl J Med 305:745, 1981. 13. Murphy GE: The clinical management of hysteria. JAMA 247:2559, 1982. 14. Herman SP, Stickler, GB and Lucas, AR: Hyperventilation syndrome in children and adolescents: Long term follow-up. Pediatr 67:183-187, 1981.