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When behavioral intervention fails: a single case report of fluoxetine's successful treatment of selective mutism

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  • Save Clinical M edicine and Diagnostics 2013, 3(1): 6-10 DOI: 10.5923/j.cmd.20130301.02 When Behavioral Intervention Failed: A Single Case Report of a Successful Treatment of Selective Mutism Using Fluoxetine Poling Bork1,*, Donna Snyder2 1Department of Graduate Studies, Brock University, St. Catharines, L2S 3A1, Canada 2Pediatrician, Niagara Falls, L2E 6G1, Canada Abstract This art icle demonstrates the remed iation of selective mut ism (SM) with fluo xetine treat ment on a male (9 years 2 months), fo llowing unsuccessful behavioral and psychosocial interventions. Although it is premature to consider pharmacological treat ment as a more effective treat ment than behavioral or other psychotherapeutic modalit ies for persistent SM, the results from this study are consistent with prior research demonstrating the efficacy of pharmacological intervention, specifically fluo xetine, in the treat ment of SM. Keywords Select ive Mutism, Behavioral, Psychosocial, Pharmacological Intervention, Fluo xet ine 1. Introduction Selective mutism (SM) is a childhood condition that is characterized by the persistent failure to speak in specific social situations such as school, despite speaking to immed iate family members at home 1. This failure to speak may persist into adolescence and adulthood if left untreated.2 The onset age for SM is between 2.7 years and 4.1 years,[3] and more females are affected by SM than males.[4] Due to its relatively low prevalence rates of 0.71%,[5] many health care professionals are unfamiliar with SM and therefore unable to provide proper help.[6,7] As a result, the lag time can be as long as four years before SM sufferers receive treat ment/intervention.[8] Although the etiology of SM remains controversial,[8,9] many researchers believe SM is an anxiety disorder[5,8,10] that closely relates to social phobia[6,11]and specific phobia – the fear of expressive speech.[12] The proposal of SM as a variant of an xiety disorder provides a pragmatic theoretical approach for assessment, treatment and research. Indeed, literature docu ments the successful treatment of SM as an anxiety disorder using behavioral[13] and pharmacological modalities.[14-16] Specifically, applied behavioral techniques such as self-modeling, shaping, contingency management, systematic desensitization, and stimulus fading have become the most frequently used method of intervention for SM. [17,18] * Corresponding author: (Poling Bork) Published online at Copyright © 2013 Scientific & Academic Publishing. All Rights Reserved SM has been found to be resistant to treat ment with documented interventions that extend over several years.[17] Early intervention is of paramount importance, to avoid the mutis m behavior strengthening, and becoming entrenched over time.[6, 19] In fact, few children receive intervention during early primary school years. Often appropriate treatment is delayed until after the child turns 7. As well, Schwart z and Shipon-Blu m[20] contends that only 30% to 40% of ch ild ren older than 12 who are diagnosed and treated appropriately will speak to a wide circle of schoolmates. Despite intervention, there are many SM cases that are intractable to conventional psychosocial intervention.[17] As such, med ications that are efficacious for an xiety-related disorders may be needed to target the serotonergic dysfunction (an underlying neurobiological deficit ) in children.[3] 2. Pharmacological Treatment for Selective Mutism Selective serotonin reuptake inhibitors (SSRIs) are effective in treat ing children with selective mutism because SSRIs increase brain levels of the synaptic neurotransmitter serotonin.[20] Thus, such increases in serotonin have been found to have considerable beneficial effects on various forms of an xiety.[21,22] Schwartz et al.[6] predicates that some parents and physicians are reluctant to include medication in the treatment of SM due to its possible adverse effects, yet, consideration of pharmacological therapy is warranted if a child does not make sufficient imp rovement with behavior therapy after 3 to 6 months, or the mutism causes significant impairment.[3,6] Pharmacological treat ment has Clinical M edicine and Diagnostics 2013, 3(1): 6-10 7 been found to be an effective intervention for persistent SM, which has been unresponsive to behavioral and psychosocial interventions. Specifically, fluo xet ine, a type of SSRI, appears to be the most promising pharmacological agent for SM.[6, 23] 2.1. Rationale for Treatment with SSRIs for Indi vi dual wi th Selective Mutism Fluo xetine (Pro zac©), a type of SSRIs, is frequently selected for child and adolescent anxiety disorders. [23-25] Since SM is conceptualized as an an xiety d isorder, it is most often treated pharmacologically with SSRIs.[6] As a group, SSRIs offer a more tolerable side effect profile than the tricycle antidepressants and Monoamine Oxidase Inhibitors.[3] Schwart z et al. [6] noted that SSRIs are not only effective for SM, but are considered by some child psychiatrists to be safe if used carefully. Research Un it on Pediatric Psychopharmacology Anxiety Study Group[26] has documented the safety and efficacy of SSRIs in treating children with various anxiety disorders, depression, and paediatric obsessive-compulsive disorder.[3, 27] Among SSRIs, fluo xetine (i.e., Pro zac) has been found to be effective for SM.[23,28]Carlson et al. [23] identified 12 of 21 (57%) pharmacological treat ments of SM used fluo xet ine. Most patients tolerated the med ication well, and their speaking (and or social) behavior improved significantly following the treatments. Fluoxet ine does not need to be tapered in small decrements over several weeks to avoid withdrawal syndrome (e.g ., dysinhibition, emotional lab ility, nausea, and headaches) that is caused by an abrupt discontinuation of a typical SSRI medication.[20] Further, not all SSRIs (e.g., citalopram, escitalopram, fluo xetine, fluvoxamine, paro xet ine, and sertraline) are targeted specifically for use with ch ild ren. Although literature has documented treatments of SM with paroxet ine,[15] and citalopram,[29] only fluo xet ine (Pro zac©), fluvo xamine (Luvo x©), and sertraline (Zoloft©) have been approved by the Food and Drug Administration (FDA) for use in children. [30] It is important to point out that the success of interventions in SM with an SSRI is inversely correlated with age, as it is confirmed that a younger age predicts a better outcome.[31] As well, although fluoxetine need not be tapered in small decrements to avoid withdrawal sy mptoms, evidence has suggested that this period of time "allows children to consolidate their progress and to expose themselves to the anxiety-provoking situations that previously elicited the symptom".[3] 3. The Case Study Overview This study reports on a child diagnosed with SM who was nonresponsive to behavioral and psychosocial intervention, but was successfully treated with fluo xet ine. The patient exhib ited symptoms of SM (only wh ispered to two peers) when he entered pre -school at 2 years 10 months. At the preschool level, the child was not diagnosed by the family physician with SM. Subsequently, the child entered grade one in a different school at the age of 5 years 10 months and became co mpletely mute inside the school. The child was diagnosed as suffering form SM at 6 years 3 month. A referral was made by the pediatrician, to a clin ician who had experience in treating children with SM using behavioral therapy. Using a framework similar to Dow, Sonies, Scheib, Moss, and Leonard’s[32] school-based mu ltid isciplinary intervention, an outpatient treatment strategic plan was set up using predominately behavioral techniques (i.e., systematic desensitization, stimulus fading, contingency management), and was imp lemented in and out of school involving a management team consisting of parents and teachers.[32] 3.1. Behavioural Intervention The boy’s supportive peers in school were identified. Classroom seating arrangements were made so that the child was situated away fro m the teacher – the most anxiety provoking figure, and was sitting next to someone he was comfo rtable with to foster speaking opportunities. During the first year, the boy’s mother came in each day as a “teacher’s helper” to encourage the child to wh isper to her inside the classroom. Then, due to work, instead of spending every day at school, the mother and father took turns and came in each morn ing before the class to conduct activities with the boy that would allow the opportunity for the child to practice speech inside school; starting in a private location (i.e., empty library) and slowly mov ing into the classroom. In addit ion, the peers who m the boy felt comfo rtable with were invited 2 to 3 times weekly for after school play dates at the boy’s home. Using the self-modeling approach, a tape recorder was used to tape the child’s speech to enable him to “talk” to his friends, for “show and tell” (while the boy squeezed his mother’s hand and hid behind her), and for reading evaluation purposes. Although walkie-talkies were also made availab le inside school and at home, the boy was unable to speak through them after nu merous attempts. After 12 months of behavioral intervention, the boy seemed to be mo re comfo rtable around his peers, but was still unable to use walkie-talkies or other medians (e.g., mother, telephone), to communicate with his friends. The mother consulted the clin ician and family physician about adding fluoxet ine into the treatment, which both promptly rejected due to possible adverse affects. The behavioral treatment continued for another 6 months and the boy was still not progressing. Frustrated, the mother also enrolled the child in art/play therapy, psychodynamic therapy; even hypnosis sessions were added. After 24 months of failed behavioral and psychosocial intervention, the boy began to receive pharmacological treat ment with fluo xetine by the pediatrician who diagnosed him with SM. 3.2. Pharmacological Treatment 8 Poling Bork et al.: When Behavioral Intervention Failed: A Single Case Report of a Successful Treatment of Selective M utism Using Fluoxetine The introduction of fluoxetine treat ment (1.25mg/day) was well tolerated and was gradually increased to 10 mg/day at week 21. At that time the boy was able to speak to a friend outside of school without eye contact (behind friend’s back) and co mmunicate co mfortably with 2 classmates through a walkie-talkie, in and out of school. In addition, the boy was also able to use a walkie-talkie to communicate with his classroom teacher. Significant improvements in other social situations (i.e., spoke directly to a younger cousin, ordered food at restaurants, spoke to swimming instructor) were also observed. At week 28, the boy continued to make further progress and spoke directly to 5 more friends (2 fro m school) at home, but only whispered to them inside the school. As the dosage increased to 12 mg/day at week 31, so too did the number of people the boy spoke to. Again, the boy tolerated the med ication well, and no side effect was reported. At week 36, the boy reportedly spoke into the microphone giving farewell wishes to the retiring principal in front of the whole school. Evidently, the boy no longer met the criteria for SM and was talking freely to everyone at that point. Treat ment effects were maintained at the 1-year follow-up. At the 2-year follo w-up, according to teachers, the boy reportedly was too talkat ive in the class, and was able to speak to everyone. The3-year follow-up showed the boy was no longer as talkative inside a new school, and was adjusting well wh ile SM remained in remission. 4. Implications for Pharmacological Treatment Despite successful treatment of pharmacotherapy in SM, treatment with med ication alone is ill-advised.[28] Medication should be considered when behavioral therapy shows inadequate response, when the mutis m is severe and has comorbid sympto ms, or if the child is older.[3, 6, 29]As mentioned, med ication is associated with possible adverse effects. Although no side effect was reported in this case study, common adverse effects of SSRIs include nausea, abdominal pain, sleep d isturbance, change in appetite and weight, lightheadedness, and fatigue. Other adverse effects, although uncommon, include serious depression, irritability, temper tantru ms, and mania,[20] allergic reactions to the drug and serotonin syndrome (confusion, muscle spasms, profuse diaphoresis, hyperthermia) and autonomic nervous system instability. In addition, there have been reports of epistaxis or bru ising in children,[33] suicidal ideation, [30, 34, 35]growth attenuation in children[36] as well as sudden cardiac deaths among children who are taking SSRIs.[37]As such, the American Psychiatric Association has published a guideline that children who are taking SSRIs must be monitored closely and should be seen by their primary physician weekly for 1 month, then every other week for another month.[6] 5. Limitation of the Study This study has several methodological limitations: 1) Instead of using standardized instruments to measure changes, this study used descriptions to elucidate behavioral change; 2) Pharmaco logical treat ment was conducted along with concurrent behavioral and psychological interventions, therefore it is possible that pharmacological treat ment “enhanced” the treatment effect of the psychosocial and behavioral interventions or vice versa; 3) Due to the rarity of this disorder, this study joins the majority of pharmacological treat ments of SM that are case studies.[23] Although a controlled study using a larger sample of children with SM can be difficu lt to conduct, a well designed study using standardized instruments to measure change across mu ltip le settings, and placebo-controlled, double-blind design studies that are more methodologically sound are needed to further establish the efficacy of pharmaco logical treat ment of SM. 6. Discussions Findings in this case study is consistent with the majority of pharmacotherapy treatments of SM in several ways: 1) The onset age of SM was within the range of 2.7 years and 4.1 years as reported by Garcia et al. in 2004;[3] 2) A lack of public awareness of SM resulted in the boy being diagnosed 3 years later; 3) The boy was unresponsive to conventional behavioral and other psychosocial interventions for 2 years; 4) Due to possible adverse effects the family physician and the SM clinician (a child psychologist) rejected the option of pharmaco logical t reatment after 2 years of failed intervention; 5) Fluo xetine provided a rapid resolution of SM when other methodologies failed; 6) The patient tolerated fluo xetine well with no reported side effects. Because the pediatrician was unfamiliar with t reating SM with fluo xetine, the medicat ion was initiated at the lowest dosage (1.25 mg/day) and increased very gradually throughout treatment. This is consistent with other pediatricians who ad minister fluo xetine beginning with 1.25 mg per day and gradually increasing the dose to a maximu m of 20 mg per day (Schwart z & Shipon-Blu m, 2005). In contrast, psychiatrists who have experience with SSRIs often prescribe higher doses of fluo xetine, up to 60 mg per day,[20] because children metabolize the drug faster than adults and thus require full doses of antidepressants.[38] Indeed, studies have demonstrated successful fluo xetine treatments of SM with an init ial dosage of 20 mg/day[16] and increased up to 60 mg/day at week 9[31] with minimal side effects. Due to the fact that our patient was treated very cautiously and gradually, whether or not he could have overcome SM much sooner with a stronger dosage of med ication remains unknown. Further methodology and sound research is needed to explore this approach. Despite limitations, this study adds to the current knowledge base in the efficacy of medicat ion treatment for SM. While pharmacological treat ment was conducted along with behavioral and psychosocial treat ments, and given that Clinical M edicine and Diagnostics 2013, 3(1): 6-10 9 the chronic symptom of mutism persisted several years and failed to respond to multiple intervention approaches, it is safe to conclude that the medication was at least partly, if not wholly, responsible for the changes in speech. 7. Conclusions This study demonstrates the importance of med ication treatment for children who have persistent SM and are unresponsive to psychosocial and behavioural interventions. It also demonstrates fluo xetine is an effective and well-tolerated treatment option for SM for this child. However, there are clear implications to treating SM with med ication due to the possible adverse effects, as well as the stipulation of using med ications that are not approved by the Food and Drug Admin istration for use in child ren. Therefore, it is important to make accurate judg ments to weigh the long-term benefit against the magnitude of side-effects caused by medication, and to monitor children closely when treating them with medication. REFERENCES [1] American Psychiatric Association. Diagnostic and Statistical M anual of M ental Disorders-4th ed., text revision, D SM -IV TR. Washington, DC: American Psychiatric Association, 2000. [2] Remschmidt, H., Poller, M ., Herpertz-Dahlmann, B., Henninghausen, K., & Gutenbrnner, C. “A follow-up study of 45 patients with elective mutism”, European Archives of Psychiatry and Clinical Neuroscience, vol. 251, no. 6, pp. 284-296, 2001. [3] Garcia, A. M ., Freeman, J. B., Francis, G., M iller, L., & Leonard, H. Selective mutism. In T. H. Ollendick & J. S. M arch (Eds.), “Phobic and anxiety disorders in children and adolescents: A clinician's guide to effective psychosocial and pharmacological interventions” (pp. 433-456). New York: Oxford University Press, 2004. 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M , Wiener (Ed.), Textbook of child and adolescent psychiatry (2nd ed., pp. 779-812). Washington, DC: American Psychiatric Press, 1997.

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