Gratification syndrome or benign idiopathic infantile dyskinesia misconstrued as epilepsy and movement disorders: a report of 2 cases

¹Department of Neurology, Andrainjato Universisty Teaching Hospital, Fianarantsoa, Madagascar, ²Department of Paediatric, Tambohobe Universisty Teaching Hospital, Fianarantsoa, Madagascar, ³Department of Neurology, PZAGA Universisty Teaching Hospital, Mahajanga, Madagascar, ⁴Department of Neurology, Befelatanana Universisty Teaching Hospital, Antananarivo, Madagascar

^&Corresponding author
Nomena Finiavana Rasaholiarison, Department of Neurology, Andrainjato Universisty Teaching Hospital, Fianarantsoa, Madagascar

Introduction    

Gratification syndrome in children refers to self-stimulatory behaviors that may become habitual over time. It is also known as benign idiopathic infantile dyskinesia or infantile masturbation and is classified as a non-epileptic paroxysmal movement disorder. It most commonly occurs in children between 3 months and 3 years of age, although it can occasionally be observed in school-aged children [1]. These behaviors have been reported in approximately 90-94% of males and 50-60% of females. Due to frequent misdiagnosis, affected children often undergo multiple unnecessary investigations, including blood tests, metabolic screening, abdominal ultrasound, gastrointestinal radiography, cerebrospinal fluid examination, skull X-ray, brain imaging, pyelography, and even invasive procedures such as cystoscopy, vaginoscopy, and proctoscopy under general anesthesia. In some cases, antiepileptic medications are inappropriately prescribed. Recognition of the clinical features of gratification syndrome is therefore essential to avoid misdiagnosis, unnecessary investigations, and inappropriate treatment [2,3]. Clinicians should remain vigilant, as delayed diagnosis is common due to limited awareness and the tendency to pursue extensive investigations [4]. Our objective is to describe the clinical profile and management of gratification syndrome in order to improve its recognition and appropriate treatment.

Patient and observation     

Case one: misconstrued as epilepsy

Patient information: a 2-year-old girl was brought to the Neurology Department of the University Hospital of Andrainjato, Fianarantsoa, for seizures that were not responding to treatment. The episodes were described as lying on her side facing the wall, with rocking movements of the body and hips, thigh adduction, and scissoring of the lower limbs (Figure 1). These episodes were associated with grunting and sweating. The child remained conscious during the episodes and responded when her name was called. Each episode lasted 1-5 minutes, was stereotyped, and occurred several times a day. The movements were not present during sleep, and the parents did not attempt to restrain them. Parents denied any history of sexual abuse. The child was born at term without complications and had no history of developmental delay. A diagnosis of epilepsy had initially been made, and she had been treated with antiepileptic drugs for several months before being referred due to poor response and the need for treatment reassessment. She is the second child of non-consanguineous parents in a monogamous family. Both parents are physicians. There was no family history of seizures or abnormal movements.

Clinical findings: her neurological examination was without particularity.

Timeline of current episodes: a 2-year-old girl presented with recurrent episodes described as lying on her side facing the wall, with rocking movements of the body and hips, thigh adduction, and scissoring of the lower limbs without loss of consciousness, misdiagnosed as seizures. Her physical examination, electroencephalography, and serum electrolyte levels were normal. She had been treated with antiepileptic drugs with a poor response. She was then referred to our neurology department, and after reviewing the recordings of the episodes, a diagnosis of childhood gratification disorder was made. Her parents were advised to distract the child during episodes, and antiepileptic drugs were discontinued. A significant frequency of episodes was reported during follow-up.

Diagnostic assessment and diagnosis: electroencephalography and serum electrolyte levels, performed before presentation, were normal. The parents were asked to record videos of the episodes. Review of these recordings revealed features consistent with gratification behavior. A diagnosis of childhood gratification disorder was made.

Therapeutic intervention: the parents were reassured and counseled, and they were advised to distract the child with interactive play during episodes. Antiepileptic medications were discontinued.

Follow-up and outcome of intervention: after 3 months, during follow-up consultation, parents reported a significant reduction in the frequency of episodes.

Patient perspective: the patient´s parents were satisfied to have the right diagnosis and to discontinue antiepileptic drugs.

Informed consent: informed consent was obtained from the patient´s parents for us to use the case

Case two: misconstrued as movement disorders

Patient information: a 3-year-old female child was presented to our department for episodes initially diagnosed as movement disorders that were not responding to treatment. Since the age of 9 months, she had exhibited repetitive rhythmic movements of the hips, thigh adduction, associated with scissoring of the lower limbs and grunting, without loss of consciousness (Figure 2). These movements could be interrupted by distraction, such as giving her toys (Figure 3). Each episode lasted approximately 3 minutes and occurred about twice per week. After the episodes, she would resume normal activities and continue playing. Based on these events, she had previously been diagnosed with movement disorders and was treated with Clonazepam. However, there was no improvement, and she was eventually referred to our department for further evaluation of paroxysmal movement disorders. She was born after an uneventful pregnancy, labor, and delivery. Her growth and development were normal. She is the only child in the family.

Clinical findings: physical and neurological examinations were unremarkable.

Timeline of current episodes: a 3-year-old girl presented with recurrent episodes described as repetitive rhythmic movements of the hips, thigh adduction, associated with scissoring of the lower limbs and grunting, without loss of consciousness, since the age of 9 months, misdiagnosed as paroxysmal movement disorders. Her neurological examination, electroencephalography, and serum electrolyte levels were normal. She had been treated with Clonazepam with poor response. She was then referred to our neurology department, and after review of the recordings of these episodes, a diagnosis of childhood gratification disorder was made. Her mother was advised to distract the child during episodes, and Clonazepam was discontinued. Significant reduction in the frequency of episodes was reported during follow-up.

Diagnostic assessment and diagnosis: electroencephalography and serum electrolyte levels were normal. The mother was asked to record the episodes on video. Review of the recordings showed features consistent with childhood gratification behavior. A diagnosis of childhood gratification disorder was made.

Therapeutic intervention: clonazepam was discontinued. The mother was counseled to distract the child when episodes occur by encouraging other activities.

Follow-up and outcome of intervention: during follow-up consultation, there was a significant reduction in the frequency of episodes according to the mother.

Patient´s perspective: the patient´s mother was satisfied with having the right diagnosis.

Informed consent: it was obtained from the patient´s parents for us to use the case.

Discussion     

Gratification syndrome in children is characterized by self-stimulatory behaviors that may become repetitive and habitual over time [1]. We report two cases from Madagascar involving young girls who were initially misdiagnosed-one as having epileptic seizures and the other as having a movement disorder. After clinical reassessment, both cases were correctly identified as childhood gratification syndrome.Gratification syndrome, or benign idiopathic infantile dyskinesia, or infantile masturbation, is classified as a non-epileptic paroxysmal movement disorder [1]. Parents often prefer the term "gratificationʺ (or"benign idiopathic infantile dyskinesiaʺ) rather than "infantile masturbationʺ because it carries less social stigma. This preference can create additional challenges in counseling parents and raising awareness about the condition [3,5]. Therefore, we avoid using the term "masturbationʺand instead describe it as a gratifying behavior that may evolve into a habit [6]. Self-gratification may begin when a child is left alone without engaging activities and discovers a pleasant and soothing bodily sensation and repeats the behavior. These episodes usually subside when the child becomes engaged in other activities or objects in the environment [5]. Gratification behavior in young children remains a diagnostic challenge due to its diverse presentations and the frequent absence of obvious genital involvement [3,7].

Regarding the sociodemographic profile, gratification syndrome often occurs in children between 3 months and 3 years of age, although it can also be seen in school-aged children [1]. These behaviors have been reported in approximately 90-94% of males and 50-60% of females [2,3]. In one case series, the age of onset ranged from 18 months to 7 years, with all cases occurring in females, similar to our series. This may be explained by the small sample sizes of studies and sociocultural factors, where such behaviors are considered more acceptable in males, leading to lower referral rates among boys [2]. In some cases, children undergo multiple unnecessary investigations, including blood tests, metabolic screening, abdominal ultrasound, gastrointestinal radiography, cerebrospinal fluid examination, skull X-rays, brain imaging, pyelography, and even invasive procedures such as cystoscopy, vaginoscopy, and proctoscopy under general anesthesia. Antiepileptic drugs are also frequently prescribed. Recognition of the clinical features of gratification disorder is therefore essential to prevent misdiagnosis, unnecessary investigations, and inappropriate treatment [2,3]. Although gratification disorder is relatively common in children, it can be a diagnostic challenge in early childhood if clinicians are not aware of it. Unlike in adolescents, it usually does not involve manual genital stimulation, and its clinical presentations are highly variable [6,8]. Girls, in particular, may present with posturing, grunting, limb stiffening, and rubbing against objects at the perineum without obvious self-touch [7].

Typical features include scissoring of the lower limbs, repetitive pelvic movements, thigh adduction, and rocking against objects such as the bed or furniture. These paroxysmal movements are often associated with grunting, facial flushing, irregular breathing, sweating, and sometimes apparent exhaustion. The episodes can be interrupted by distraction. These events are frequently misdiagnosed as seizures, dystonia, urinary tract infection, or infantile colic. Gratification syndrome is frequently mistaken for epileptic seizures, particularly when there is a coexisting history suggestive of epilepsy, as in our cases. Key features supporting the diagnosis include preserved consciousness throughout the episodes and their resolution when the child is distracted [1,7]. There are no specific diagnostic criteria for gratification disorder in the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5), or the International Classification of Diseases (ICD-10/ICD-11). Diagnosis is therefore based on clinical recognition and the exclusion of other conditions such as epilepsy, childhood-onset dystonia, paroxysmal movement disorders, abdominal pain, or colic. In all cases, video recording is extremely important to rule out these mimics [4]. Video documentation of the episodes is invaluable, as parents may use inaccurate or misleading terminology when describing the events. Clinicians should therefore routinely request video recordings when evaluating suspected seizures or movement disorders [8]. Typical clinical features in children, as described by Yang et al., include: i) onset between 3 months and 3 years of age; ii) stereotyped episodes of variable duration; iii) vocalizations such as quiet grunting; iv) facial flushing with diaphoresis; v) pressure on the perineum with characteristic lower limb posturing; vi) preserved consciousness; (vii) cessation with distraction; viii) normal physical examination; and ix) normal laboratory findings. Our patients exhibited most of these features [5,6,8,9].

Some authors have suggested that early exposure to sexual materials or behaviors may influence the development of masturbatory behaviors in children [2]. The perception of masturbatory behaviors in children as a taboo plays a significant role in referral patterns, with many cases being referred to neurology rather than psychiatry [2]. Treatment is important, as some children with gratification syndrome may have difficulties with self-regulation and delayed gratification, which could predispose them to impulse control disorders and deficits in executive function. Studies have reported that up to 20% of children diagnosed with gratification disorder may later develop attention-deficit/hyperactivity disorder (ADHD) [4]. Management primarily involves psychoeducation, behavioral modification, distraction techniques, parent management training, and age-appropriate sex education [4]. Parents need to understand that this is a common and generally benign behavior in children, and that most children outgrow it over time. It is important to avoid scolding or punishing the child, as this may reinforce the behavior and negatively affect self-esteem. Instead, parents should gently guide the child and explain the need to avoid such behaviors, particularly in public settings and progressively in private contexts as well [8]. Referral to a behavioral psychologist may be beneficial for some families [1]. In cases where gratification disorder does not respond to behavioral interventions, pharmacological treatment may be considered. Some studies have reported the use of selective serotonin reuptake inhibitors and antipsychotic medications. However, behavioral interventions combined with psychoeducation remain the first-line management [2,7,10].

Conclusion     

Gratification behavior is a normal aspect of psychosexual development in children; however, it should always be considered as a differential diagnosis. Clinicians need to be aware of this condition to avoid misdiagnosing it as epilepsy, movement disorders, or other conditions. Video recording of these episodes is particularly helpful in establishing the correct diagnosis in neurological practice. Accurate diagnosis can prevent unnecessary investigations and inappropriate treatments, reducing both the medical burden and parental anxiety. Parental reassurance and behavioral management are effective in reducing the frequency of episodes and addressing associated stigma

Competing interests     

The authors declare no competing interests.

Authors' contributions     

Patient management: Nomena Finiavana Rasaholiarison, Odilon Rahamefy Randrianasolo. Data collection: Nomena Finiavana Rasaholiarison, Odilon Rahamefy Randrianasolo. Manuscript drafting: Nomena Finiavana Rasaholiarison, Prudence Ramamonjinirina, Parany Haja Rabearisoa. Manuscript revision: Lala Andriamasinavalona Rajaonarison, Julien Razafimahefa and Alain Djacoba Tehindrazanarivelo. All authors approved the final version of the manuscript.

Figures     

Figure 1: patient lying on her side facing the wall, with thigh adduction and scissoring of the lower limbs (case 1)

Figure 2: patient with thigh adduction and scissoring of the lower limbs (case 2)

Figure 3: ceasing of the episode when the patient is distracted with a balloon

References