Childhood gratification syndrome: Demystifying the clinical conundrum with a narrative literature review of the past 5 decades

Abstract

Background:

Childhood gratification syndrome (CGS) refers to self-stimulatory or masturbatory behaviors in children, which may have an onset as early as in infancy (IGS).

Aim:

The aim of this review is to understand the various clinical manifestations of CGS/IGS and their clinical differentiation from commonly misdiagnosed neurological and physical illnesses and to formulate a preliminary approach to their diagnosis and management.

Methods:

This narrative review is based on a search of literature over the past 50 years (1972–2022) in three online databases (PubMed/Medline, Embase, and Google Scholar).

Results:

The behaviors are episodic, occurring for brief periods, involving posturing, stereotypical limb movements, pubic pressure with autonomic hyperactivity, and postepisodic lethargy. They mimic seizures, movement disorders, abdominal pain, and tics. The paper also highlights the gap in the current knowledge to guide future research in the area. CGS usually represents nonpathological “pleasure-seeking” habits of childhood, but at times, it may become problematic for the child and his family. A careful history and videotape analysis of the events confirms the diagnosis and behavioral therapy with parental reassurance as the mainstay of treatment.

Conclusion:

A better understanding and clinical awareness of the CGS are necessary to prevent misdiagnosis and delay in appropriate intervention.

INTRODUCTION

Masturbation is self-stimulating one’s genitalia for sexual pleasure.[1] Although historically considered a social taboo, it is now considered a normal practice among adults and often done until orgasm is reached. Almost 65% of adult males and 40% of adult women report masturbating at some point in their life.[2]

Masturbatory behaviors are not uncommon in children,[3,4] although they can be quite different from adult practices with a relative lack of actual genital stimulation. Therefore, masturbatory behaviors in children have been addressed more appropriately as ‘childhood gratification syndrome (CGS)’. At times, the behaviors may involve stroking the genitals with fingers, hands, or everyday objects like toys, often associated with twitching of lower limbs for several minutes with or without jitteriness at other parts of the body, diaphoresis, or facial flushing.[5,6] Due to their episodic and transient nature, they are often mistaken as neurological events.[7] An understanding of this otherwise normal physiological behavior is therefore important to prevent unnecessary investigations and interventions. This work intends to describe and comprehend the construct through a narrative review of the existing literature since the past 50 years to help guide future research.

MATERIALS AND METHODS

A literature search on the topic during the past 50 years (1972–2022) was performed in databases (PubMed/Medline, Embase, and Google Scholar) using keywords and Boolean operators with a search string ‘(Infantile gratification) OR (childhood gratification) OR (infantile masturbation) OR (childhood masturbation)’. The search strategy returned 90 potential papers, from which 50 relevant articles with no duplicates and full text available in English have finally been included in this work. The relevance of the articles was determined after reviewing their titles and abstracts by at least two authors independently. Articles not reporting the desired topic were removed, while minor disagreements were adressed through meetings. The retained articles were read in full to extract the desired data/information.

RESULTS

Most available articles on the topic in the past 5 decades are case reports and cross-sectional studies [Figure 1]. The detailed summary of the articles is given in Table 1.

Figure 1.

Bibliography review (year 1972–2022) showing the types and number of studies included here

Table 1.

Summary of studies on Childhood Gratification Syndrome in the past 50 years (1972-2022)

Study; Study type (N)		Findings/Results
		Gender, Presenting age (Age of onset)		Clinical features		Management		Outcomes/Remarks
Leung et al., 1993[1]; NR		Prevalence: M>F Commonest age of onset: 4y		genital stimulation against an object with muscle tension, rhythmic body movements, associated pelvic thrust and autonomic changes.		The need for open discussion on sexuality between parents and children, age appropriate sex-education and greater social interaction and play in children were highlighted.
Cacciatore et al., 2020[3]; CSS (n=507)		Finnish daycare professionals reported verbal and behavioral expressions of sexuality in children (1–6 years): The commonest topics were body (curiosity, exploring the functions etc.) and emotions (infatuation, tenderness toward others). 71% professionals open masturbation by children.
Hansen and Balslev, 2009[4]; CS (n=13)		M (n=2), F (n=11); 6.1m*		Fisting, grasping of inanimate objects or mother’s hand, chorea-like ‘piano playing’ finger movements, hand pressure or bimanual manipulation of genitalia; cessation on distraction.		Video-recording of the events helped in final diagnosis.		Role of video-recording in diagnosis was highligted.
Dhaher et al., 2020[5]; CS (n=44)		M (n=22), F (n=22), 3.2±0.41y* (3.2±0.8y)*		Rubbing the genitals with hands or an object in prone or supine posture with flushing, diaphoresis and sleep; nocturnal in 20% cases; variable frequency, mean duration: 5 mins.		Initial diagnosis: unidentified (455%), neurodermatitis (31.8%), UTI (13.6%), diaper dermatitis (9%); Reassurance of patients once CGS is confirmed.		Social embarrasment was a major cause for referral.
Yang et al., 2005[6]; CS (n=12)		F (n=12), (11m)*		Dystonic posturing, rhythmic pelvic movements, diaphoresis; ceasation on distraction		Normal neurological examinatios; video-recording of the events helped in final diagnosis.		Direct observation of the events was emphasised. While parental psychoeducation was useful, scolding and punishing the child was counterproductive.
Phillips and Seshia, 2013[7]; Editorial		Various aspects of CGS with respect to its epidemiology, etiology, clinical manifestations, differential diagnosis and management were discussed.
Ajlouni et al., 2010[8]; CCS (n=26)		M (n=3), F (n=10), 2.67±1.28y* (19.5m)!		Mastusbatory behavior mostly in prone or supine posture with facial flushing and friction between thighs; frequency: 3–20 episodes/day; median duration: 3.9 mins.		Normal neuroimaging in all, abnormal EEG in 1 out of 8 children tested. Serum estradiol levels were significantly lower (P=0.03) in cases versus controls.		Reduced estradiol levels were associated with CGS.
Jan et al., 2013[9]; Cohort study (n=19)		M (21%), F (79%), (7m)*		Self-stimulatory behaviors (pressure on perinium with autonomic features) in wakeful states, duration: 5–30 mins. 21% cases later developed ADHD.		74% cases were misdiagnosed as epilepsy or movement disorder; thorough history, video-recordings, unremarkable developmental or neurological findings and normal EEG helped diagnosing CGS; parental reassurance and BT was helpful with remission by age 1–3y (mean 1.9 y).		A link between CGS and subsequent ADHD was hypothesised.
Nechay et al., 2004[10]; RA of chart (n=31)		M (n=11), F (n=20), (median age of onset: 10.5m)		Dystonic posturing (n=19), grunting noises (n=10), rocking (n=9), eidetic imagery (n=7), sweating (n=6); occuring in sleep (n=5); median frequency: 7 episodes/week; median duration: 2.5 mins		Median age of diagnosis: 24.5m; median delay in diagnosis: 11m; differentials considered: epilepsy (n=21), dystonia and pain abdomen; video-recording helped confirm diagnosis (n=9).		CGS is an important consideration in nonepileptic paroxysmal events in children. Video-recordings allow confident diagnosis.
Wolf and Singer, 2008[11]; NR		Description of CGS along with its various differentials (abdominal pain, dystonia, epilepsy, dyskinesias) with emphasis on direct observation of the behavior and reassurance to family members in management. The term ‘masturbation’ has been referred to as distressing for parents.
Fleisher and Morrison, 1990[12]; CS (n=5)		F, 7-27m (>2m)		Stereotyped posturing with pressure on the pubic or suprapubic area, associated quite grunting, diaphoresis and autonomic features; absence of genital rubbing or copulatory actions; episodes lasting from 1 min to hours with no loss of responsiveness.		Initial misdiagnosis as abdominal pain or seizures led to unnecessary investigations. Later prohibition of punishments, parental psychoeducation and behavioral therapy for the child were found useful.		Several factors stimulating CGS (sexual abuse, family stress, perineal discomfort and parental guilt feeling) were discussed.
Murat et al., 2022[13]; CSS (n=124)		M (23.8%), F (76.3%), 45.5m* (24.16m)*		Lower socioeconomic status, less education of the parents, unplanned pregnancy, parents playing less with children, parents cosleeping with children, sleep problems and history of UTI in the child were factors significantly associated with CGS.
Unal, 2000[14]; Cohort study (n=50)		M (n=16), F (n=34), 48.7±24.5m*		At 2 years follow-up: 78% of the children completely stopped the masturbatory behaviors. Those who did not fully stop were significantly younger, began masturbating earlier and had more masturbatory frequencies than others.
Izadi-Mazidi and Riahi, 2020[15]; CSS (n=600)		M (44.6%), F (55.4%), 5y 7m*		65 (10.8%) children had pathological CGS; commonly observed behaviors: gental manipulation (58.46%), genital rubbing on inanimate objects (18.46%), and both (6.15%); preferred time: anytime (33.84%), bedtime (12.30%), when alone (15.38%); 23% were reportedly bored before the event.		ADHD with anxiety, tics, communication disorder, learning disorder, OCD or mood disorders were commonest commorbidity (32.30%); parental awareness, thorough history and video-recordings helped correct diagnosis.		Maternal education and parental awareness influenced childhood sexual behaviors and reporting of the events.
Deda et al., 2001[16]; CR (n=1)		M, 3.5m (3.5m)		Crossing of thighs with tonic posturing while waking up from sleep or during sleep with no loss of consciousness; 6–9 events daily each lasting 15-72s.		Hospitalization for observation; initially diagnosed as infantile spasm but EEG were normal; referred to psychiatry and later lost to follow-up.		Careful history and investigations are keys to diagnosis.
Wilkinson and John, 2018[17]; CR (n=1)		F, 1.5y (1y 5m)		Touching the vaginal area when off diaper; distractable during the episode.		No hitory of diaper rash, vaginal discharge or any genital lesions. Reassurance was helpful.		The differentials (genitourinary disorders, seizures, abdominal colic and psychological disorders) were discussed alongside various masturbatory manifestations of CGS.
Al-Mendalawi, 2011[18]; Letter		Three comments on the study by Ajlouni et al., 2010[8] were made: a) regarding the reference ranges of pediatric sex hormones studied in the paper, b) regarding the sensitivity and specificity of the sex hormone measurement methods, and c) regarding the need for extensive studies for generalisation of the interesting findings of the study.
Couper and Huynh, 2002[19]; CR (n=1)		F, 4y (1y)		Sitting down and clutching the abdomen above the pubis with facial flushing, events lasting for 15 min, without loss of awareness; angersome behavior if interrupted.		Video-recording of the events helped diagnosis of CGS; supportive conseling was useful.		A link between CGS and gonadotrophic surge in first few months of infant life was hypothesised. Freudian proposition on sexuality and hysteria was discussed.
Chen et al. 2015[20]; RA of VEEG (n=81)		M (n=19), F (n=12), 10.5m*		Motor (leg extension/adduction, hands between legs), behavioral (starring or arrest of activity) and autonomic (flushing) features; occuring in awake state with intact responsiveness; duration: <30s.		38 PNEEs were recorded in 31 children. CGS was diagnosed in 10.5% of the cases.		PNEEs can frequently coexist with epilepsy.
Doust et al., 2016[21]; CCS (n=623)		M: F::1:7, <12y		Masturbatory behavior in prone or lateral decubitus position.		Urine microscopy was positive for mucus in 99.94% of the children with CGS opposed to 8.3% of the children without CGS. Urinary mucus correlated significantly (P<0.001) was with masturbatory behavior.		Normal EEG during episodes, no response to antiepileptics, typical video-recording findings, and detailed history help diagnosing CGS.
Unal, 2000[22]; CCS (n=104)		M: F::1:2, 53.5±32.3m* (32.4±23.8m)*		Children with CGS had increased sleep difficulties (P<0.001) and shorter breast-feeding (P<0.001) compared to controls. An inciting event (weaning, sibling birth or separation of parents) or a genitourinary disorder was associated with the onset of CGS.
Fernandes et al., 2016[23]; CR (n=1)		M, 32w POG		USG abdomen of mother showed fetal penile manipulation associated with penile tumescence.		-		First demonstration of fetal in-utero masturbation.
Meizner, 1987[24]; Letter (n=1)		M, 28w POG		On routine USG: the fetus was observed to grasp his penis and make masturbatory movements for 15 min.		-		First description of in-utero fetal masturbation.
Franić and Franić, 2011[25]; CR (n=1)		F, 1y 2m (9m)		Crossing over of legs in a sitting posture with alternate contraction		Normal clinical examination and EEG. Behavioral modification and		CGS though benign, may cause
				and relaxation of thigh muscles; associated rocking pelvic movements, facial flushing, grimacing, accelerated breathing and diaphoresis.		addressal of parental distress helped improvement.		great distress to the families which needs adequate addressal.
Moktan et al., 2021[26]; CS (n=3)		F, 3y (1.5y); F, 4y (1y); F, 8y (7y)		Case 1: Rubbing genitalia on a hard object while standing. Case 2: Rocking movement of body and genitalia against floor while in prone position along with thrusting hip movements. Case 3: Rhythmic pelvic thrusts with arching backward and stroking genitals on a hard surface. All cases: normal awareness and responsiveness during episodes, ceasation on distraction; frequency: 4-5/week to 20/day; duration: 3-30 mins.		In 2 children episodes of UTI precipitated the event. Distraction, ABC analysis followed by behavioral management helped remission.		Secondary causes like perineal irritation may present with CGS-like behavior. Possible precipitating and predicposing factors of CGS were discussed.
Omran et al., 2008[27]; CS (n=3)		F, 10m (2m); F, 2y (3m); F, 2.5y (8m)		Contraction, extension and scissoring of lower limbs with autonomic changes; associated body rocking and leg rubbing.		Seizure was ruled out after thorough history, clinical examination and investigations (EEG, CT, Blood and urine workup)		Points of difference between CGS from epilepsy were discussed.
Wulff et al., 1992[28]; CS (n=2)		F, 5m (5m); M, 6m (6m)		Hips and knees flexion, leg crossing, clenched fingers, head turned to one side with autonomic features; responsive during episodes.		Ruling out other differentials, AEDs were unhelpful, reassurance was helpful.		Frequencies of the episodes reduced over next 6m.
De Alwis et al., 2006[29]; CR (n=1)		M, 4y (3y)		Touching, scratching and shaking genitalia along with arching of back, neck twisting and rhythmic pelvic movements; 5-12 episodes daily, each lasting for 5 min with autonomic features.		Seizure was ruled out. USG abdomen revealed a 1.5 cm bladder calculus which was removed by vesicolithotomy, followed by dramatic improvement.		Emphasis on the need to evaluate for genitourinary disorders in CGS was given.
Livingston et al., 1975[30]; CR (n=1)		F, 7y (3y 4m)		Lying on abdomen with stiffened posture and rocking movement in relative privacy; often having a staring gaze and profuse sweating with some drowsiness postepisode.		Detailed investigations (neuroimaging, EEG, CSF analysis, etc.) were normal, was initially diagnosed as epilepsy and started on AEDs, later a careful history helped a diagnosis of CGS.		The differential diagnoses were discussed.
Mink and Neil, 1995[31]; CR (n=1)		F, 3y 5m (1y)		Unusual posturing of right upper and lower limbs with right tilt of her head and occasional shaking of right foot; seemingly during which she looked inattentive but responsive; event frequency: 5-6/day; duration: 5 mins to several hours.		Extensively evaluated by a neurologist and geneticist; Serum biochemistry, electrolytes, hemograms were normal; normal EEG; MRI brain revealed a nonsignificant small supracellar cyst; sexual abuse was ruled out; a video-recrding of the event helped in diagnosis.		The authors discussed other differentials like PDC and seizures, and emphasized CGS is just another deviation of a normal childhood behavior.
Otaigbe, 2008[32]; CR (n=1)		F, 15m (3m)		Unusual rocking with thigh adduction (at 3m age); forward bending and rocking on hard surface (at 10m age); associated lip smacking, eye rolling and shaking.		Video-recording helped in the diagnosis. Physical examination revealed a slightly hyperemic clitoris possibly due to rubbing behavior. Counceling, proper clothing and diaper use helped remission.		The relatives of this child intended genital mutilation as per their cultural practices but the authors stressed the role of supportive therapy.
Bradley, 1985[33]; CR (n=1)		F, 5.5y (5m)		Leaning forward on an object with facial flushing and labored breathing.		Extensive gastrointestinal and genitourinary evaluation, parental psychoeducation.		Frequency decreased a year later.
Arafat and Khan, 2019[34]; CR (n=1)		F, 6y (4.5y)		3-4 episodes per week, lasting for 5 mins, characterised by rubbing the pubic area on hard objects.		Unremarkable physical examination and MSE., Treatment involved distraction and reinforcement to the child and parental psychoeducation with supportive therapy.		The role of psychiatrist in managing CGS in a ‘sexually conservative’ country was highlighted
Kaya et al., 2012[35]; CR (n=2)		MZ twins: F, 11m (8.5m), F, 11m (9.5m)		Flexion of legs toward abdomen and rubbing together; associated groaning and autonomic features; cessation on distraction.		Diaper dermatitis was ruled out. Video-recording helped in the diagnosis. Treatment constituted behavioral therapy.		Possibile role of shared genetics in development of CGS around the same time in the twins.
Nemati et al., 2022[36]; SR (n=5)		Arpiprazole and escitalopram were described to be effective in decreasing the behaviour of CGS. Psychoeducation and reassurances were effective for parents. Distraction and reinforcement techniques were effective for children.
Mallants and Casteels, 2008[37]; NR		Prevalence: F>M		Episodes of stereotyped posturing of lower limbs with perineal pressure, autonomic changes for variable duration; intact consciousness; cessation on distraction.		Psychoeducation and supportive therapy.		Close differentials (dyskinesia, paroxysmal dystonia, epilepsy and abdominal pain) were discussed.
Långström et al., 2002[38]; CSS (n=649)		MZ compared to DZ twin pairs had higher prevalence of problematic child masturbatory behavior. Genetics influenced masturbatory behavior significantly (77%, 95% CI=9–96%), apart from family environment and stressful events like sexual victimisation. Both genetic and socioenvironmental factors were shown to a role in CGS.
Thabit et al., 2021[39]; CSS (n=876)		M (n=3), F (n=9), 42m*		Nonepileptic seizures (NESs) were diagnosed in 19.5% of the children, of which 7% received a diagnosis of CGS (1.4% of the total; n=12), with a female preponderance.
Mills and Hedderly, 2014[40]; Editorial		Discusses the differentials for movement disorders in childhood including CGS.
Aslan and Gungor, 2022[41]; RA of VEEG (n=87)		Long-term vEEG images and reports of 87 children admitted with a diagnosis of epileptiform activity were analyzed: CGS was diagnosed in 6.5% of the children following vEEG monitoring.						vEEG: goldstandard in differentiating PNEEs from epilepsy.
Bodensteiner and Sheth, 2006[42]; Letter		The authors stressed the importance of home video-recordings as an objective evidence to diagnose CGS. This should be accompanied by a checklist for the parents so that they do not miss any details of the paroxysmal event. They also suggested a systematic evaluation of these recordings akin to that of a neuroimaging for better understanding the event.
Casteels et al., 2004[43]; CS (n=5)		F (n=5), <1y		Self-stimulatory behaviors.		Video-recording of the events helped diagnosis of CGS. Parental reassurance waned of distress and anxiety in the parents.		The role of video-recording in the diagnosis was emphasised.
Nagy and Hollody, 2019[44]; CS (n=5)		F, 2.5y (2.5y)		2 week history of ‘weird’ movements during sleep: turning to one side, crossing her legs with scissoring movements lasting for 20-30 mins; frequency 2-3 times/day; patient was reactive during the event.		Video-recording of the event helped in the diagnosis of CGS.		Differences from epileptic events were discussed.
Omranifard et al., 2013[45]; RCT (n=90)		Risperidone (0.25-1 mg/day) added to usual BT over 4w resulted in a significant reduction in the mean frequency of CGS (from 2.7±0.9 to 1.1±0.5 times/day) in comparison to the control (only BT) group (from 2.6±0.9 times/day to 1.4±0.6 times/day).
Albertini et al., 2006[46]; CR (n=1)		M, 5y		Compulsive masturbatory behavior in addition to clinical features of autism.		CARS score of 41 suggested severe autism. IQ assessed was 81. EEG was normal. MRI brain revealed bilateral perisylvian polymicrogyria. Mirtazapine 5 mg/day helped significant reduction in masturbatory episodes and other behavior abnormalities.		Role of mirtazapine in compulsive sexual behaviors (here CGS) and symptoms of autism were highlighted.
Indian Studies
Maqbool et al., 2017[47]; CR (n=1)		M, 13y (13y)		Insertion of finger in anus to get rid of constipation leading to shame, guilt, stinky fingers and rectal bleeding.		-		Possible differentials including manipulation of prostrate for pleasure was discussed. A link with Freudian anal stage of psychosexual development was discussed.
Biswas et al., 2020[48]; Cohort study# (n=54)		M (n=25) F (n=29) (20.98±9.31m)		Commonest presentations: rocking in prone position (44.44%), intermittent dystonic posturing with crossed legs (42.59%), direct genital manipulation (5.56%); episodes usually occurred in solitude and boredom; mean frequency: 10.31±5.10/week; mean duration: 9.83±5.98 mins.		2 children were initially misdiagnosed as having epilepsy. Diagnosis was reached from history, home video-recordings and direct observation of events during hospital stay.		CGS should be considered as a differential in cases of epilepsy and childhood paroxysmal events. Home video-recordings are useful in diagnosis.
Dudipala et al., 2021[49]; Cohort study (n=19)		M (n=2), F (n=17), 20m* (17m)*		Commonest presentations: scissoring (63.15%) and leg twisting (57.89%); ceasation on distraction; mean frequency: 4 episodes/day; mean duration: 4 mins.		Initially diagnosed as seizures (31.75%) on AED, UTI (10.05%) on antibiotics and infantile colic (26.31%) on probiotics and antispasmodics. EEG and neuroimaging normal in tested cases. After revised diagnosis, only parental reassurance was given.		Misdiagnosis leads to unnecessary investigations, pharmacological treatments with their side-effects and delay in referral to psychiatrist.
Biswas et al., 2020[50]; CS# (n=3)		M, 1y 8m (1y 6m); M, 3y 8m (2y 8m); F: 3y 5m (2y 9m)		Direct genital manipulation with penile erection in one case, sweating, anger on interruption, termination of events on distraction.		Normal physical and neurological examination. Symptoms improved with distraction and behavioral therapy.		CGS may rarely present with direct genital manipulation.
Pandurangi et al., 2016[51]; CR (n=1)		F, 2y 2m (2y 2m)		3-4 daily episodes (5-10 mins) featuring manual rubbing of the genitalia in prone position with autonomic features.		Normal physical examination and EEG. Treatment constituted reassurance, parental counseling behavioral therapy to the child. Remission in next 4m.		A relapse after an year again responded well to behavior therapy.
Babu, 2012[52]; Letter		Discussion on the various manifestations of CGS and how it can be misdiagnosed as seizures, dystonis, abdominal pain etc., A high degree of suspicion and video-recording of events by parents using smartphone camera can help establish the diagnosis. Child abuse must always be ruled out.

*Mean±standard deviation of the parameter of the cases/study population has been provided as available, !median age, y: years, m: months, w: weeks, mins: minutes, s: seconds Study designs: Letter: Letter to the editor, CR: case report, CS: case series, RA: retrospective analysis, CSS: cross-sectional study, CCS: case control study, NR: narrative review, SR: systematic review, RCT: randomised clinical trial, POG: period of gestation (intra-uterine), CGS: childhood gratification syndrome (used synonymously to infantile/childhood masturbation) PNEE: paroxysmal nonepileptic event, AED: antiepileptic drugs, MSE: mental status examination, IQ: intelligence quotient, MRI: magnetic resonance imaging, MZ: monozygotic, DZ: dizygotic, CI: confidence interval, UTI: urinary tract infection, EEG: electroencephalography, EEG: electroencephalography, vEEG: video-EEG, CT: computed tomogram, USG: ultrasonography, PDC: paroxysmal dystonic choreoathetosis, ADHD: attention-deficit/hyperactivity disorder, CARS: childood autism rating scale, BT: behavioural therapy

Nosology

Penile erection during sleep or genital manipulation and vaginal secretion upon stimulation is common in neonates but disappears in a few months.[53] In contrast, childhood masturbatory behaviors are active self-stimulation of genitalia in a prepubescent child and may be reported as early as in infancy.[1] It has been usually addressed as ‘gratification disorder’ and ‘infantile/childhood masturbation’.[8,9,10] The former is more acceptable to the parents due to the associated taboo with the latter.[10,11] But these terms fail to describe the stereotypical activities that bring the child to medical attention. The term ‘benign idiopathic infantile dyskinesia’ was suggested since the presentations lacked any underlying dysfunction in basal ganglia and related structures.[10] Later, a more precise alternative, ‘paroxysmal hyperkinetic motor syndrome’, was proposed.[7] However, the self-stimulatory behaviors have a more psychological than organic basis and referring them as ‘disorders’ undermines their nonpathological nature. The synonymous ‘childhood gratification syndrome (CGS)’ encompasses both the varied clinical presentations and the normalcy of the involved behaviors.

Etiology

Self-stimulatory behaviors in children were earlier considered to be motivated by their “erotic feelings”.[54] Freud, in his psychoanalytic theory, discussed about “libido”, the sexual energy underlying one’s pleasure-seeking urges. He proposed that since birth, there is an innate tendency to seek pleasure through stimulation of the “erogenous zones” of the body, which are postinfancy centered around the perineum.[47,54] His conceptualization of the “latency period” from age six till puberty also explains the relative decline in masturbatory acts in children during this period. Martinson explained that “infantile sexuality” is a result of the more general “pleasure-pain principle”.[7] Elkin, however, rejected the interpretations of childhood self-stimulatory behaviors based on “adult sense”. Masturbation in adults involves a “complex interplay of emotions, attitudes, and fantasies” involving a “physical, cognitive and emotional maturity” that a child lacks. In case of a child, it may be a simple pleasurable act becoming a habit.[7] Jean Piaget described habit formation in infants of 4 months through coordinated integration of actions like sucking and grasping.[55] Interestingly, self-stimulatory actions in children have an onset around this age. The behaviors are possibly discovered randomly while playing but are reinforced by their pleasurable effects.[7] Therefore, they are part of a spectrum that includes thumb-sucking, body-rocking, and other similar childhood activities performed for comfort.[6]

For older children, excessive masturbation may be a result of familial discord and lack of physical attachment with parents.[12] Self-stimulation was reportedly higher in children with parents spending less time with them.[13] An inciting event like weaning, birth of a sibling, or separation of the parents was found to precede the onset of CGS.[14] Evidently, childhood self-stimulation was found to occur during boredom, anxiety, loneliness, or excitement.[15] Other social factors associated with CGS were lower socio-economic status, less education of the parents, unplanned pregnancy, and shorter breast-feeding.[13,14] Furthermore, parental attitudes toward sexuality may influence sexual behaviors in children with higher reports of CGS from families facilitating honest disclosure about the issue.[15] Sexual abuse also predisposes children to masturbatory behaviors and must be probed for in every case.[12,16] The child’s feelings of “vulnerability” or “guilt” associated with the actions and frustration following parental interruptions were seen to reinforce the behaviors.[12]

Perineal discomfort due to local irritation or infection (e.g., diaper rash) may intensify a child’s sensations and precipitate CGS. Similarly, genitourinary diseases, urinary tract infections (UTIs), perineal surgeries, and constipation may exacerbate such behaviors.[14,15,17]

A study found lower levels of serum estradiol in children diagnosed with CGS but lacked generalizability due to a small sample size.[8,18] Also, a surge in luteinizing hormone/follicle stimulating hormone (LH/FSH) during infancy alongside increasing motor sophistication has been implicated for the development of the autoerotic behaviors, particularly in girls.[12,19] A higher prevalence of comorbid psychiatric illnesses, such as attention-deficit/hyperkinetic disorder (ADHD), tics, learning disorders, obsessive-compulsive disorder (OCD), sleep problems, anxiety, and mood disorders was reported among children with CGS.[13,14,15] Furthermore, at least 20% of a small cohort of children with self-stimulatory behaviors developed ADHD in the long term, suggesting a possible link between ADHD and CGS.[9] Additionally, episodes of self-stimulatory behaviors mimicking seizures may coexist with epilepsy, making diagnosis difficult.[20]

Phenomenology

Infantile masturbation was first described by Still in 1909.[6] Since then, several case reports and series have tried exploring the phenomenon with variable success. However, despite high individual differences in the presentations, Yang et al.[6] discussed certain common features in the onset, duration, semiology, and clinical findings. Expanding upon his findings, the clinical characteristics of CGS are enumerated below:

1. Female preponderance: The male: female ratio across studies has ranged from 1:9 to 1:2.[13,21,22] The sex difference may be attributed partly to the high gonadotrophic activity in girls during their early life months[19] and partly to the anatomical differences and sociocultural factors.[12,22]

2. Early onset: Masturbatory movements have been observed in the fetus using ultrasonography.[23,24] Nevertheless, studies have shown the onset of CGS following a bimodal peak, one around age 4 and another during adolescence.[6] In the former group, the behaviors usually developed between 3 months and 3.5 years.[6,8,10,13,22,48]

3. Rigid posture with genital manipulation: A typical childhood masturbatory episode involved dystonic posturing with pressure around the perineum.[6,10,12] There was associated scissoring of limbs, forward bending, or rocking of the pelvis.[25,26,27,28,49] Direct stimulation of the genitalia using hands or rubbing against hard surfaces was observed in others.[5,49,50] The playful genital manipulation along with contractions of muscles of the pelvis, extremities, and back mimicked the ‘orgasms’ of a masturbating adult.[56]

4. Associated stereotyped behaviors: Typically, five distinct hand activities (fisting, grasping, chorea-like piano-playing finger movements, genital manipulation, and bimanual handling of items) have been described in CGS.[4] Other movements reported during the episodes were lip-smacking, eye-rolling, staring, jitteriness, and neck twisting.[29,30,31,32] The behaviors were replicable in particular settings, like in a car seat and during boredom or anxiety.[6,15]

5. Autonomic features: The events were associated with ‘quiet grunting’, facial flushing, labored breathing, and profuse sweating.[5,8,10,19,20,25,30,33,50] Nonspecific changes (tachycardia and high-amplitude waves) were recorded on EEG.[1] After an episode, the child usually felt tired and tended to fall asleep akin to the experience of orgasm in adults, a trance state followed by general relaxation and lethargy.[1] However, it may be re-emphasized that genital manipulation, reaching orgasm, or autonomic features are not always present in CGS.

6. Variable frequency and duration: Reportedly, the episodes had a frequency of occurrence from 3/week to 20/day,[8,34,51] each lasting from a few seconds to hours (average 4–5 minutes).[5,12,20,49] The wide range may be explained by sample heterogenicity across the reports and individual variations given the nature of the phenomenon.

7. Intact consciousness and responsiveness: An episode could abruptly be ceased by distracting the child[4,35,36,49] but would make him angry and frustrated.[19] Normal awareness, ability to recall the events, responsiveness, and distractibility are clinically most important differentiating features from seizures.[19]

Defining a strict boundary between normal and deviant behaviors in context of CGS is difficult. However, given the wide variability in presentations, it is worth exploring the symptoms of CGS dimensionally on a spectrum, with episodic stereotyped behaviors (with or without genital stimulation) on one end and dysfunctional masturbatory behaviors on the other. Clinically, problematic CGS is characterized by excessive or obsessive masturbations or age-inappropriate self-stimulation in public.[37,38]

Physiological changes

Autonomic hyperactivity is clearly appreciable clinically during a typical episode similar to the masturbatory findings in adults.[1] But the distinct stages of libido-arousal-orgasm-resolution, as seen in adults, may not be appreciable in children. Also, in an adult male, there is an appreciable elevation of plasma levels of pregnenolone and dehydroepiandrosterone, among several other sex hormones, following a masturbatory act.[1] However, contrastingly, in children, significantly lower levels of estradiol levels have been found during masturbation.[8] The repeated involvement in the behavior and display of displeasure, if interrupted, hints at the possible role of pleasure centers being activated during the act.

Differential diagnoses and assessments

Diagnosis of CGS can be difficult in view of its striking similarity with many neurological constructs. Nechay et al. (2004)[10] reviewed 31 cases where 21 of the patients were previously misdiagnosed with seizure disorder. Thabit et al. (2021)[39] in a recent study on 876 children observed nonepileptic seizures in 19.5% of cases, of which 7% had CGS. Unlike in seizures, a child with CGS can be distracted during an event. Also, seizures are abrupt and spontaneous, while self-gratifying behaviors are relatively slow to start and voluntary. An epileptic seizure may occur anytime and anywhere, whereas childhood masturbatory activities are usually performed in one’s comfort or privacy.[10] However, there was one case where automatisms of absence status manifested as compulsive masturbation.[1] Similarly, 11 of the 12 cases reported by Yang et al.[6] were initially referred to the Neurology for movement disorders. Paroxysmal dystonic choreoathetosis (PDC) can occur unilaterally or bilaterally without any precipitant. But, unlike in PDC, a child with CGS can “abruptly and easily” abort an episode.[6] The repetitive movements in CGS may also appear like motor tics but lack the preceding sensorimotor phenomenon.[40] They may be mistaken as motor stereotypies, but stereotypes are fixed, have less variability, and usually involve upper-limb movements with relative absence of genital stimulation and autonomic arousal.[40] Also, local pathologies like abdominal pain, perinitis, vulvovaginitis, and UTI can make a child take the assumed posture due to the related discomfort. “Retentive posturing” associated with perennial tightening, grunting, and facial flushing in children who withhold stools may be confused with self-stimulation.[12] Also, the possible history of sexual abuse needs to be ruled out in each case.[16]

CGS may be confused with other childhood conditions involving repetitive behaviors, like intellectual disability (ID), autism spectrum disorder (ASD), and compulsive sexual behaviors (CSBs). Stereotyped behaviors are present in 34–69% of children diagnosed with ID and over 95% of those with ASD.[57] An executive dysfunction with ‘impaired generative ability’ together with ‘automatic perceptual reinforcement’ has been proposed as the basis of stereotypy in ID and ASD.[57,58] Clinically, ID presents with impaired intellectual and adaptive functioning and ASD with added deficits in social-emotional reciprocity and communication.[59] Moreover, in ASD, the repetitive behaviors are typically ritualized, are restricted to fixed interests, and exhibit hyper- or hyporeactivity to sensory inputs.[58,59] Due to social, communication, and sensory issues, some individuals with ASD may show hypersexuality or non-normative sexual behaviors. In contrast to CGS, these individuals are often older (adolescents) and may experience heightened sexual desires, orgasms, and even paraphilic fantasies.[60,61] Similarly, CSB usually affects adolescents and is characterized by intense preoccupation with sex, extensive pornography use, and risky sexual behaviors.[62]

A major concern in the management of CGS is diagnostic delay (median 11 months), attributable to the relative lack of clinical awareness, numerous referrals, and unwarranted investigations.[10] In certain cases, a gap of more than 3 years between symptom onset and final diagnosis was reported.[19,30,33] Thus, the understanding of CGS as a normal physiological entity needs consideration prior subjecting a child to a plethora of laboratory procedures and pharmacological interventions with their side effects.[49]

As such, the roles of EEG, neuroimaging, and neurological examinations are limited to ruling out organic pathologies with no diagnostic implication for CGS.[41] Most authors highlighted the importance of homemade videotapes of the events in assessing and confirming the diagnosis as episodes are rare in controlled clinical settings.[4,9,12,21,42,43,44,48,52] Fleisher and Morrison described 12 characteristic features pertaining to history, description of the event, and course to help the diagnosis.[12] CGS may be suspected in a typical case with unremarkable birth and developmental history, normal neurological and clinical examinations, relevant video-recording findings, and nonsignificant EEG and neuroimaging reports [Figure 2].

Figure 2.

Approach to a child presenting with episodes of self-stimulatory behaviors. CGS: childhood gratification syndrome (used here synonymously with infantile/childhood masturbation), vEEG: video electroencephalogram, PDC: paroxysmal dystonic choreoathetosis, CSA: child sexual abuse, UTI: urinary tract infection, ID: intellectual disbility, ASD: autism spectrum disorder, CSB: compulsive sexual behavior, BT: behavioral therapy, CBT: cognitive behavioural therapy, SSRIs: serotonin reuptake inhibitors

Treatments and prognosis

A diagnosis of CGS can be distressing to the family, especially in a conservative society.[11,34] Therefore, destigmatization is crucial to reduce the perceived stigma and encourage treatment approval. Normalization and understanding that the symptoms are not a result of poor parenting or intentional misbehavior prevent the formation of any guilt or shame. Subsequently, parental reassurance, psychoeducation, and behavioral training of the child remain the mainstay of therapy.[25] Yang et al.[6] recommended redirecting the child’s attention toward engaging in activities with positive reinforcements instead of threats or punishments. Such efforts at habit reversal focusing on replacing the problematic self-gratifying behaviors with compatible alternatives are useful in reducing the frequency and intensity of the symptoms. Additionally, a healthier attitude toward sexuality, sexual counseling, and moral education can help in the positive development of the growing child.[1]

A single randomized control trial (RCT) found combined risperidone (0.25–1 mg/day) and behavioral therapy (BT) to significantly reduce the episodes compared to BT alone.[45] Also, a systematic review showed aripiprazole and escitalopram to be effective in reducing the behaviors of CGS.[36] Mirtazapine has been reported to be useful in a single case study.[46] However, pharmacotherapy may be reserved for cases where the masturbatory behaviors are problematic, dysfunctional, and refractory to psychotherapeutic approaches. Unlike such scenarios, self-stimulation in most children is a normal psychological behavior. Normal childhood masturbation does not interfere with sexual practices in adulthood,[1] and along with age, the behaviors usually subside.

Indian studies and cross-cultural comparisons

Six studies could be identified from India: two cohort studies, two case reports, one case series, and one letter to the editor [summarized separately in Table 1]. The two prospective observational studies showed similar findings: female preponderance, symptoms onset between 3 months and 3.5 years, diagnosis between 4 months and 4 years, and episode frequency from 4/day to 10/week.[48,49] The usual presentations were intermittent scissoring and twitching of legs or rocking in the prone position, ceasing on distraction.[48,49] Direct genital manipulation was not uncommon, sometimes accompanied by penile erection and sweating.[48,50,51] Misdiagnosis was common, with one study reporting an initial diagnosis of epilepsy in 31% of children and infantile colic in 26%.[49] Diagnosis of CGS was reached from history, normal neurological examinations, home video tapes, and hospital observations.[48,50,51] In most cases, parental reassurance and behavioral therapy were effective.[48,49,50,51]

Despite the limited number of Indian studies, not many differences were evident compared to the observations from other parts of the world. The cross-cultural indifference, particularly female majority and similar onset and presentation, may be explained by an underlying genetic or neurobiological cause, minimally influenced by one’s culture or environment. This hypothesis potentially explains higher rates of neurodevelopmental disorders in these children. Cultural factors may still impact how the symptoms are perceived, diagnosed, and treated. Alternatively, self-stimulatory behaviors could be a natural part of development, discovered during random activities and reinforced by their pleasurable sensations. This can still explain the higher occurrence of CGS in children with ADHD, ID, and ASD as they often exhibit difficulties in sensory integration/processing. CGS as a learned behavior seems more probable, given its benign long-term course and positive outcomes with behavioral therapies. It is also possible that both neurobiology and development might contribute together to the origin of CGS.

Future avenues for research

Though uncommon, research on CGS has been very limited. Many features, like the varied symptomatology, lack of pleasure, and prolonged (for several hours) episodes in some cases, need explanation.[7] Few reports have indicated high association of ADHD with CGS, but the role of psychiatric illnesses in the development of CGS needs further exploration. Additionally, future studies may aim to understand the long-term course, effects on sexual behaviors in adulthood, and the underlying neurobiological basis. Cross-country multicentric studies are needed to identify the sociocultural and religious factors influencing the presentations.

Strengths and limitations

This review provides a comprehensive summary of the available literature on CGS published over the past 5 decades from three large databases. Furthermore, a comparison between the findings in the Indian and foreign studies has beeen provided to better understand the similarities in the presentation and cultural influences. The key findings can help enhance clinical awareness about CGS and prevent diagnostic delays and unnecessary investigations or medications. However, being a narrative review, it is limited by lack of systematic data synthesis and potential bias due to subjective interpretations. Also, additional literature which may be found in other databases has been missed.

CONCLUSIONS

CGS involves self-stimulatory/masturbatory behaviors in a child, which are normal but may be distressing for the parents. At times, CGS may become problematic and dysfunctional necessitating treatment. It is most often mistaken with seizures, movement disorders, abdominal pain, and tics subjecting the child to unnecessary investigations and treatments. Careful history and videotape analysis are important for diagnosis, and parental reassurance and behavioral therapies are the mainstay of treatment. Clinical awareness can prevent delay in diagnosis and intervention, while further research is needed for understanding the varied presentations, neurobiological underpinnings, and sociocultural influences.

Keypoints

• Self-stimulation in children (childhood gratification syndrome, CGS) is mostly nonpathological.

• CGS, a clinical conundrum mimicking neurological events, is misdiagnosed very often.

• Careful history and videotape analysis of the events confirm the diagnosis.

• Behavioral therapy and parental reassurance are the mainstay of treatment.

• More research is needed looking for future neurobehavioural trajectories of children exhibiting gratification syndrome.

Data availability statement

Data sharing is not applicable to this article as no new data were created or analyzed in this study.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.