- Email: [email protected]
CITALOPRAM FOR HEAD-BANGING
LETTERS TO THE EDITOR
psychiatrists as well as pediatricians is that it is rarely considered in the differential diagnosis of unexplained physical symptoms, In part this appears related to the difficulty physicians (child psychiatrists included) have in believing that a parent could be reporting symptoms that either are nonexistent or occur as a result of a parent’s actions. I have recently been involved in such a case in which a child had dose to 80 hospital admissions for a variety of unexplained physical symptoms (including failure to thrive, apnea, fevers of unknown origin) before the possibility of Munchausen by proxy was even considered. 1 would urge all my colleagues, especially those who consult with pediatricians and those involved in somatization disorders, to include this in their differential diagnoses when they are dealing with children with multiple hospitalizations. Allex Weintrob, M.D. Cornell University Medical College New York
Campo JV,Janscn-McWilliams MS, Comer DM. Kcllchcr KJ (1999). Somatization in pediatric primary care: association with psychopathology. functional impairment, and usc of services. ]Am Arad Child Ah&sr P s y r h i u ~38:1093-1 101
Note: See the related Clinical Perspectives in this issue,
p. 668.
CITALOPRAM FOR HEAD-BANGING
To the Editor: Head-banging is one of a number of stereotypic symptoms that are not uncommon in children with normal intelligence (Sallustro and Atwell, 1978) and that are extremely prevalent in children with mental retardation (Hyman et al, 1990). Although the natural course of head-banging in children with normal intelligence is often benign (Abe et al, 1984), it may also be associated with significant injury and may meet DSMIVcriteria for a stereotypic movement disorder. The use of behavioral treatments for head-banging has been documented, and there is a smaller body of literature on the use of serotonergic, dopaminergic, opioid, and other agents in stereotypic movement disorder in adult patients with normal intelligence (Stein and Simeon, 1998). We would like to report the successful use of a selective serotonin reuptake inhibitor (SSRI) for head-banging in a child with normal intelligence. A 5-year-old boy was referred for treatment of head-banging. The head-banging had begun 2 years before, and it now occurred almost every day, typically 5 or 6 times a day, until bleeding occurred. The patient’s developmental history indicated that normal milestones had been achieved, and there was no history of head injury. His h i l y history was positive for attention-deficidhyperactivitydisorder (ADHD) and seizure disorder in one brother. O n clinical interview the patient
544
was found to meet DSM-Ndiagnostic criteria for stereotypic movement disorder, ADHD, and enuresis. A physical examination (including ear examination), routine laboratory investigations, EEG, and brain magnetic resonance imaging all proved normal. A neuropsychologid assessment was obtained, and the report stated that on the Griffiths Mental Development Scale, the patient had average intellectual development, strengths in gross motor coordination, difficulties in fine motor coordination, adequate speech and language development, and impaired problem-solving and perceptual organization skills, all consistent with a diagnosis of ADHD. A 1-month trial of methylphenidate (15 mg p.0. daily), together with parental counseling, had been commenced by the referring physician, but during that time increased symptoms of ADHD were reported. A trial of carbamazepine had subsequently also been undertaken without effect. In view of the diagnoses of ADHD and enuresis, a trial of imipramine (25 mg at night) was initiated in our clinic. Over the next several months there was an improvement in both ADHD and enuresis, but no change in head-banging. Imipramine was replaced by citalopram 20 mg daily. During the next 2 weeks, the head-banging ceased. The patient‘s ADHD symptoms remained improved, but enuresis returned. The fdmily elected to continue the citalopram for the following year, during which time the head-banging remained in remission. O n the basis of both preclinid and clinical studies, several authors have suggested the importance of the serotonin system in the mediation of self-injurious behavior (Stein and Simeon, 1998). Certainly, our finding that imipramine was ineffective and citalopram effective in the treatment of head-banging is consistent with previous work indicating that serotonin reuptake inhibitors may be selectively effective in the treatment of repetitive self-injurious symptoms in adults with stereotypic movement disorder (Castellanos et al., 1996), trichotillomania, and onychophagia. In view of the favorable adverse effect profile in comparison with other agents used for self-injurious and stereotypic behaviors, the SSRIs may be well worth considering as part of the management of such symptoms. Wendy Vogel, M.B. Department of Psychiatry University of the Witwatersrand Johannesburg, South Africa Dan J. Stein, M.B. MRC Research Unit on Anxiety Disorders University of Stellenbosch Tygerberg, South Africa K, Oda N, h a t o m i M (1984),~~~~d history and +.,ificance of head-banging, head-rolling and breath-holding spclls. Dev Med Child Nnrml26614-648 Cvtellanos FX, Ritchie GF, Marsh WL, Rappon JL (1996).DSM-IVstercorypic movcmcnt disordcr: pcrsistencc of stcreotypies of infancy in intellectullly normal adolescents and adults.]CIin Psychthy 5 7 116-122
J . A M . A C A D . C H I L D A D O L E S C . PSYCHIATRY, 3 3 : 5 , MAY 2000
LETTERS TO T H E E D I T O R
Hyman SL, Fisher W, Mercugliano M, Cataldo MF (1990), Children with self-injurious behavior. Pcdizh.irr 85:437-441 Sallustro F, Aovcll CW (1978), Body rocking, head banging and head rolling in normal children. 1Pcdiun 93:704-708 Stein DJ, Simcon D (1998). Pharmacotherapy of rtcrcorypic movement disorders. Pychian Ann 28~327-334
RISPERIDONE IN PRADER-WILL1 SYNDROME
To the Editor: We would like to report our promising results with risperidone in the treatment of patients suffering from Prader-Willi syndrome (PWS). First described in the German literature in 1956, PWS is defined as a genetic disorder that can appear in a sporadic form. Typical behavioral symptoms of PWS include stubbornness, impulsivity, aggressiveness, explosivity, temper tantrums, self-mutilation, poor peer relationships, obsessive food-seeking, and hyperphagia. The behavioral manifestations of PWS, which are severe in nature, become prominent in late childhood and adolescence and may lead to morbid obesity accompanied by lifethreatening cardiac-respiratory complications. To date, PWS and its behavioral manifestationslack a proven and efficient management modality. Because of the behavioral disturbances, various modes of psychotropic medications have been suggested, among them serotonin reuptake inhibitors, stimulants, lithium, anticonvulsant medications, and classic neuroleptics (Fava, 1997). Typical antipsychotic medications are sometimes recommended in view of their efficacy in treating aggression and hostility (Cmbor et al., 1995). However, in view of the sedative effect of typical neuroleptics and the particular vulnerability of PWS patients to extrapyramidal side effects, even at low dosages, it seems that this medication category is unwarranted in these young nonpsychotic patients. Because of the limitations of the aforementioned options, we decided to conduct a clinical trial with risperidone, a benzisoxazole atypical antipsychotic medication with a high affinity to serotonin 2A and dopamine D2 receptors. Our hypothesis that risperidone might prove beneficial in the treatment of PWS patients was based on its observed beneficial effect in autistic patients with disruptive behavioral manifestations and pervasive developmental disorders and on the assumption that the pathogenesis of PWS, like that of autism, involves dopamine and serotonin systems (McDougle et al., 1998). Our decision to choose risperidone for the treatment of PSW was additionally motivated by findings indicating that PWS patients share similar genetic coding with autistic patients and by a recent report suggesting risperidone to be superior to classic neuroleptics (haloperidol) and better tolerated in children and adolescents aged 5.5 to 16 suffering from autism, Aspergeis disorder, and disintegrative or other pervasive developmental disorders (McDougle et al., 1997). O n the basis of the substantial clinical improvement in autistic patients after risperidone treatment (Horrigan and Barnhill, 1997), we postu-
J . A M . A C A D . C H I L D A D O L E S C . PSYCHIATRY, 3 9 : 5 . MAY ZOO0
lated that risperidone might also prove efficacious in treating PWS. We posited that a favorable response to risperidone among PWS patients might constitute evidence that dysregulation of central dopamine and serotonin (5-hydroxytryptamine) neurotransmitters, already shown in autism, plays a role in both disorders and that the effectiveness of risperidone stems from its being a balanced serotonin-dopamine antagonist. Our clinical trial of risperidone treatment was conducted on 3 patients (aged 18-21) suffering from PWS after they failed to respond to other acknowledged modes of treatment. Scores on the Clinical Global Impression (CGI) scale and the Overt Aggression Scale (OAS) and weight were obtained during 2 baseline visits and again after 15 and 37 weeks of treatment with risperidone. Risperidone proved to be beneficial in all 3 PWS patients. Low dosages (1-2 mg/day) of risperidone brought about notable clinical improvement with no apparent adverse side effects. All of the measures evaluated--general behavior, CGI, OAS, and weight-reacted favorably to the treatment protocol. In particular, communication with the patients became much easier due to improvement in their behavior in general; they were less aggressive, less impulsive, and less prone to outbursts of anger, and control and management of their obsessive food-seeking behavior became easier. Their tendency to lie and steal with respect to food subsided. Perhaps the most surprising impact of the risperidone regimen was that 2 of the patients (weighing 77 and 89 kg) actually lost weight (9.5 and 12.5 kg, respectively). The third patient gained weight (4 kg) because of the special dietary program necessitated by a recent biliary-pancreatic bypass operation. All 3 patients found it easier to maintain a balanced diet. Most reports on risperidone treatment of behavioral disturbances cite weight gain as a main side effect. The patients subjected to this clinical trial lost weight along with, or as a direct result of, the general improvement in their behavior problems which, as noted, center on obsessive food search and intake. We suggest that because risperidone provides a partial answer to the core symptom of PWS, namely extreme food compulsion, the overall net result is weight reduction rather than the weight gain usually expected with risperidone intake. It is recommended that further clinical research involving more subjects and longer follow-up periods be conducted to substantiate the efficacy of risperidone in treating patients suffering from PWS. Rimona Durst, M.D. b y Rubin-Jabotinsky, M.D. Sergey Raskin, M.D. Gregory Katz,M.D. Josef Zislin, M.D. Jerusalem Mental Health Center Kfar Shad Hospi~al Hebrew University-Hadassah Medical School Jerusalem
545
psychiatrists as well as pediatricians is that it is rarely considered in the differential diagnosis of unexplained physical symptoms, In part this appears related to the difficulty physicians (child psychiatrists included) have in believing that a parent could be reporting symptoms that either are nonexistent or occur as a result of a parent’s actions. I have recently been involved in such a case in which a child had dose to 80 hospital admissions for a variety of unexplained physical symptoms (including failure to thrive, apnea, fevers of unknown origin) before the possibility of Munchausen by proxy was even considered. 1 would urge all my colleagues, especially those who consult with pediatricians and those involved in somatization disorders, to include this in their differential diagnoses when they are dealing with children with multiple hospitalizations. Allex Weintrob, M.D. Cornell University Medical College New York
Campo JV,Janscn-McWilliams MS, Comer DM. Kcllchcr KJ (1999). Somatization in pediatric primary care: association with psychopathology. functional impairment, and usc of services. ]Am Arad Child Ah&sr P s y r h i u ~38:1093-1 101
Note: See the related Clinical Perspectives in this issue,
p. 668.
CITALOPRAM FOR HEAD-BANGING
To the Editor: Head-banging is one of a number of stereotypic symptoms that are not uncommon in children with normal intelligence (Sallustro and Atwell, 1978) and that are extremely prevalent in children with mental retardation (Hyman et al, 1990). Although the natural course of head-banging in children with normal intelligence is often benign (Abe et al, 1984), it may also be associated with significant injury and may meet DSMIVcriteria for a stereotypic movement disorder. The use of behavioral treatments for head-banging has been documented, and there is a smaller body of literature on the use of serotonergic, dopaminergic, opioid, and other agents in stereotypic movement disorder in adult patients with normal intelligence (Stein and Simeon, 1998). We would like to report the successful use of a selective serotonin reuptake inhibitor (SSRI) for head-banging in a child with normal intelligence. A 5-year-old boy was referred for treatment of head-banging. The head-banging had begun 2 years before, and it now occurred almost every day, typically 5 or 6 times a day, until bleeding occurred. The patient’s developmental history indicated that normal milestones had been achieved, and there was no history of head injury. His h i l y history was positive for attention-deficidhyperactivitydisorder (ADHD) and seizure disorder in one brother. O n clinical interview the patient
544
was found to meet DSM-Ndiagnostic criteria for stereotypic movement disorder, ADHD, and enuresis. A physical examination (including ear examination), routine laboratory investigations, EEG, and brain magnetic resonance imaging all proved normal. A neuropsychologid assessment was obtained, and the report stated that on the Griffiths Mental Development Scale, the patient had average intellectual development, strengths in gross motor coordination, difficulties in fine motor coordination, adequate speech and language development, and impaired problem-solving and perceptual organization skills, all consistent with a diagnosis of ADHD. A 1-month trial of methylphenidate (15 mg p.0. daily), together with parental counseling, had been commenced by the referring physician, but during that time increased symptoms of ADHD were reported. A trial of carbamazepine had subsequently also been undertaken without effect. In view of the diagnoses of ADHD and enuresis, a trial of imipramine (25 mg at night) was initiated in our clinic. Over the next several months there was an improvement in both ADHD and enuresis, but no change in head-banging. Imipramine was replaced by citalopram 20 mg daily. During the next 2 weeks, the head-banging ceased. The patient‘s ADHD symptoms remained improved, but enuresis returned. The fdmily elected to continue the citalopram for the following year, during which time the head-banging remained in remission. O n the basis of both preclinid and clinical studies, several authors have suggested the importance of the serotonin system in the mediation of self-injurious behavior (Stein and Simeon, 1998). Certainly, our finding that imipramine was ineffective and citalopram effective in the treatment of head-banging is consistent with previous work indicating that serotonin reuptake inhibitors may be selectively effective in the treatment of repetitive self-injurious symptoms in adults with stereotypic movement disorder (Castellanos et al., 1996), trichotillomania, and onychophagia. In view of the favorable adverse effect profile in comparison with other agents used for self-injurious and stereotypic behaviors, the SSRIs may be well worth considering as part of the management of such symptoms. Wendy Vogel, M.B. Department of Psychiatry University of the Witwatersrand Johannesburg, South Africa Dan J. Stein, M.B. MRC Research Unit on Anxiety Disorders University of Stellenbosch Tygerberg, South Africa K, Oda N, h a t o m i M (1984),~~~~d history and +.,ificance of head-banging, head-rolling and breath-holding spclls. Dev Med Child Nnrml26614-648 Cvtellanos FX, Ritchie GF, Marsh WL, Rappon JL (1996).DSM-IVstercorypic movcmcnt disordcr: pcrsistencc of stcreotypies of infancy in intellectullly normal adolescents and adults.]CIin Psychthy 5 7 116-122
J . A M . A C A D . C H I L D A D O L E S C . PSYCHIATRY, 3 3 : 5 , MAY 2000
LETTERS TO T H E E D I T O R
Hyman SL, Fisher W, Mercugliano M, Cataldo MF (1990), Children with self-injurious behavior. Pcdizh.irr 85:437-441 Sallustro F, Aovcll CW (1978), Body rocking, head banging and head rolling in normal children. 1Pcdiun 93:704-708 Stein DJ, Simcon D (1998). Pharmacotherapy of rtcrcorypic movement disorders. Pychian Ann 28~327-334
RISPERIDONE IN PRADER-WILL1 SYNDROME
To the Editor: We would like to report our promising results with risperidone in the treatment of patients suffering from Prader-Willi syndrome (PWS). First described in the German literature in 1956, PWS is defined as a genetic disorder that can appear in a sporadic form. Typical behavioral symptoms of PWS include stubbornness, impulsivity, aggressiveness, explosivity, temper tantrums, self-mutilation, poor peer relationships, obsessive food-seeking, and hyperphagia. The behavioral manifestations of PWS, which are severe in nature, become prominent in late childhood and adolescence and may lead to morbid obesity accompanied by lifethreatening cardiac-respiratory complications. To date, PWS and its behavioral manifestationslack a proven and efficient management modality. Because of the behavioral disturbances, various modes of psychotropic medications have been suggested, among them serotonin reuptake inhibitors, stimulants, lithium, anticonvulsant medications, and classic neuroleptics (Fava, 1997). Typical antipsychotic medications are sometimes recommended in view of their efficacy in treating aggression and hostility (Cmbor et al., 1995). However, in view of the sedative effect of typical neuroleptics and the particular vulnerability of PWS patients to extrapyramidal side effects, even at low dosages, it seems that this medication category is unwarranted in these young nonpsychotic patients. Because of the limitations of the aforementioned options, we decided to conduct a clinical trial with risperidone, a benzisoxazole atypical antipsychotic medication with a high affinity to serotonin 2A and dopamine D2 receptors. Our hypothesis that risperidone might prove beneficial in the treatment of PWS patients was based on its observed beneficial effect in autistic patients with disruptive behavioral manifestations and pervasive developmental disorders and on the assumption that the pathogenesis of PWS, like that of autism, involves dopamine and serotonin systems (McDougle et al., 1998). Our decision to choose risperidone for the treatment of PSW was additionally motivated by findings indicating that PWS patients share similar genetic coding with autistic patients and by a recent report suggesting risperidone to be superior to classic neuroleptics (haloperidol) and better tolerated in children and adolescents aged 5.5 to 16 suffering from autism, Aspergeis disorder, and disintegrative or other pervasive developmental disorders (McDougle et al., 1997). O n the basis of the substantial clinical improvement in autistic patients after risperidone treatment (Horrigan and Barnhill, 1997), we postu-
J . A M . A C A D . C H I L D A D O L E S C . PSYCHIATRY, 3 9 : 5 . MAY ZOO0
lated that risperidone might also prove efficacious in treating PWS. We posited that a favorable response to risperidone among PWS patients might constitute evidence that dysregulation of central dopamine and serotonin (5-hydroxytryptamine) neurotransmitters, already shown in autism, plays a role in both disorders and that the effectiveness of risperidone stems from its being a balanced serotonin-dopamine antagonist. Our clinical trial of risperidone treatment was conducted on 3 patients (aged 18-21) suffering from PWS after they failed to respond to other acknowledged modes of treatment. Scores on the Clinical Global Impression (CGI) scale and the Overt Aggression Scale (OAS) and weight were obtained during 2 baseline visits and again after 15 and 37 weeks of treatment with risperidone. Risperidone proved to be beneficial in all 3 PWS patients. Low dosages (1-2 mg/day) of risperidone brought about notable clinical improvement with no apparent adverse side effects. All of the measures evaluated--general behavior, CGI, OAS, and weight-reacted favorably to the treatment protocol. In particular, communication with the patients became much easier due to improvement in their behavior in general; they were less aggressive, less impulsive, and less prone to outbursts of anger, and control and management of their obsessive food-seeking behavior became easier. Their tendency to lie and steal with respect to food subsided. Perhaps the most surprising impact of the risperidone regimen was that 2 of the patients (weighing 77 and 89 kg) actually lost weight (9.5 and 12.5 kg, respectively). The third patient gained weight (4 kg) because of the special dietary program necessitated by a recent biliary-pancreatic bypass operation. All 3 patients found it easier to maintain a balanced diet. Most reports on risperidone treatment of behavioral disturbances cite weight gain as a main side effect. The patients subjected to this clinical trial lost weight along with, or as a direct result of, the general improvement in their behavior problems which, as noted, center on obsessive food search and intake. We suggest that because risperidone provides a partial answer to the core symptom of PWS, namely extreme food compulsion, the overall net result is weight reduction rather than the weight gain usually expected with risperidone intake. It is recommended that further clinical research involving more subjects and longer follow-up periods be conducted to substantiate the efficacy of risperidone in treating patients suffering from PWS. Rimona Durst, M.D. b y Rubin-Jabotinsky, M.D. Sergey Raskin, M.D. Gregory Katz,M.D. Josef Zislin, M.D. Jerusalem Mental Health Center Kfar Shad Hospi~al Hebrew University-Hadassah Medical School Jerusalem
545