This prospective cohort study was conducted among children presenting to the outpatient clinic in the Department of Paediatric Medicine, Burdwan Medical College, Burdwan, West Bengal, India from January 2014 to December 2019. The study protocols were approved by the Ethics Committee of Burdwan Medical College, Burdwan (vide memo no. BMC/PG/719). Informed consents were taken from parents, before the beginning of the study. Everyone had the right to withdraw from the study at any time. No added cost was imposed on the families and all information was retained as confidential.

Inclusion criteria: All children 5 years or below who were diagnosed to have the condition during the period mentioned were included in the study.

Exclusion criteria: Children in whom the diagnosis of epilepsy was confirmed, those with abdominal conditions like Gastro-Oesophageal Reflux Disease (GERD) or abdominal colic, documented Urinary Tract Infection (UTI), children with genital irritation (diaper rash, pin worms, poor perineal hygiene) were excluded.

Since there is no firm guideline in literature till date regarding how to diagnose gratification behaviours in children, diagnostic criteria were formulated based on evaluation of the consistent clinical findings observed in previous studies [5,13-15]. Children who fulfilled the following two criteria were assumed to have gratification behaviour or masturbating behaviour:

Diagnosis was made on the basis of careful history taking and watching videoclips meticulously in presence of both the parents. Wherever video-recording of the event was unavailable or was nonconclusive, the child was admitted for 48 hours to directly observe the episode in order to make a correct diagnosis.

A data collecting sheet was developed which included information on demographic characteristics, clinical presentation, investigations and modes of treatment. Demographic data included: birth history (birth order, mode of delivery, birth weight, important perinatal events if any), immunisation status, breast feeding practices, major past illness, family structure, education and occupation of parents. Details of gratification spells were noted like- age of onset and diagnosis, frequency, duration, situations when the episodes were observed, behaviours during the spells, physical changes noticed during the episodes and appearance of the child before and after those episodes. Records were kept regarding investigations performed and wherever treatment was required.

Statistical data were initially entered into Microsoft Excel spreadsheet and later they were imported to Statistical Package for the Social Sciences (SPSS) software. All data were analysed by simple descriptive statistics using SPSS 26.0 software (IBM Corporation, USA).

A total of 54 patients were included in the study. Among them, 29 (53.70%) were girls and 25 (46.30%) were boys [Table/Fig-1].

Twenty-eight children (51.85%) in the present study were first born children of their parents. All children in the study group (n=54) were born by institutional delivery. Immunisation status of the study population was 100% as per national immunisation schedule. Twenty-three children (42.59%) received some extra vaccines as per recommendation of Indian Academy of Paediatrics from child care physicians. No major adverse health events in their children were reported by 49 families; three children had history of simple febrile seizure and two had history of neonatal jaundice treated successfully by phototherapy alone [Table/Fig-1].

Thirty-two parents (59.26%) have had come with other medical problems and then, one of the parents declared the concern during conversation. Eighteen parents (33.33%) came directly to seek advice on the peculiar posture/behaviour of their children. Four children (7.41%) were referred to us by local paediatrician for further evaluation. Among these, two children (3.70%) were diagnosed to have seizure disorder, were put on anticonvulsant and patients were not responding. Two patients (3.70%) were referred for further work up of dystonic posturing [Table/Fig-2].

It was possible to make a firm diagnosis by careful history taking and observation of videoclips in 49 children (90.74%). In spite of careful examination of video-recordings, diagnosis was in doubt in 2 of such children. Three families could not provide any video-recording of the events. Thus, 5 children (9.26%) required hospitalisation so that the episodes could be directly observed by the medical personnel to make a more confident diagnosis [Table/Fig-2].

The frequency of events varied from 3/week to 19/week (mean of 10.31±5.10 times/week). The mean length of events was 9.83±5.98 minutes (range 3 min to 26 min) [Table/Fig-2].

Parental aggression was noticed in one case (1.85%) and familial disharmony was noticed in two cases (3.70%). History of gratification habit in other sibling was reported in two families.

Detailed physical, neurological and genital examination were performed on all children and findings were mostly noncontributory. Routine urine and stool tests and urine culture results, performed on all, were reported normal. MRI brain and EEG were performed on two children referred to us as epilepsy. No abnormality was detected in any, and anticonvulsant was stopped in both. Parents of all children were advised to use distraction technique on noticing the spell and to avoid creating stressful situation for them. This simple step helped most of the children (n=38, 70.37%) in the present study group. Sixteen children (29.63%), however, were sent for behaviour therapy and all of them responded well within next one year. No other behavioural problem was noticed among them.

Masturbation is a normal human behaviour and it can occur at any age [1,2,6]. Most of the physiological changes that happen in adolescents or adults, also happen to these children. The difference between children doing it and others is that, children do not know what they are doing is sexual yet [1-3].

Among 54 children in the present study population, 29 (53.70%) were girls and 25 (46.30 %) were boys. Most of the studies in literature shows higher prevalence of gratification behaviours among girls [Table/Fig-3] [4,5,11,13-15]. Tashakori A et al., however found boys masturbating more than girls in their study on 98 children [9]. Most parents in the present study (n=32, 59.26%) did not come to seek medical attention with masturbation or gratification as principal complaint. They came with some other health issues of their children and during conversation; one of the parents initiated the topic. Almost all the parents expressed a feeling of guilt and shame while discussing these events. Othman SA and Unal F also had similar experiences during their study period [11,16]. Exact prevalence of childhood gratification and its’ gender distribution is very difficult to know since lot of factors like cultural and religious beliefs, social awareness and understanding regarding sex and sexuality, and educational status of the parents, all interfere with the pattern of reporting of such events across different cultures, societies and countries [14,17].

Most of the children in the present study manifested such behaviour before 3 years of age (mean age at first presentation was 20.98±9.31 months). Nechay A et al., observed a mean age of 12.5 months at presentation in their study [5]. Hiyam Shamo’on found in his study on Jordan population, 18.5 months as mean age at first symptoms [14]. Nechay A et al., found in their study of 31 children with gratification habits: average age at diagnosis was 35 months; the frequency of events varied from 1/week to 12/day; mean length of the spells was 9 minutes [5]. Hiyam Shamo’on reported the frequency of the event to vary from 3 times/week to 10 times/day [14]. The mean duration of the event was 7 minutes. The study findings on these features were consistent with these studies [5,14]. Average delay between first manifestation and diagnosis of the event was less in the present study (mean delay of 5.3 months, range- 1 month to 1 year 1 month) as compared to Nechay A et al., study findings (mean delay of 16 months) [5]. Most common patterns of gratification noticed in the present study were tightening of the thighs with dystonic posturing in girls (n=23, 42.59%) and rocking in prone position in boys (n=17, 31.49%). Direct genital manipulation was uncommon and was seen only in 3 children (5.56%). These findings were consistent with other studies [5,7,11,14]. In most studies children seemed to have a delightful feeling during the episodes [4,5,11,14]; Nechay A, et al., however, reported one child to be cyanosed and frightened during the spell [5]. Two children in the present study population were initially misdiagnosed as epilepsy and were put on anticonvulsants, and other two children were referred to us for further evaluation of possible dystonia. Misdiagnosis in this age group possibly happens due diverse and sometimes peculiar manifestations of the spells, striking absence of direct genital manipulation in most cases and lack of awareness among parents and paediatricians about this entity. Nechay A et al., Fleisher DR and Morrison A, and Ibrahim A and Raymond B reported instances of childhood gratification being initially misdiagnosed and treated as epilepsy [5,7,10]. Mink JW and Neil JJ, and Yang ML et al., reported that childhood gratification was misdiagnosed as movement disorders [12,13]. Fleisher DR and Morrison A, and Couper RT and Huynh H have described dramatic examples of masturbation mimicking abdominal pain in girls [7,8]. With smartphones being available to almost every family now-a-days, it is possible to video-record all these events. This can clarify most of the findings and misdiagnosis can thus be avoided.

Aetiology and predisposing factors of gratification behaviours in children are still poorly understood [9,15,17,18]. Possible sexual abuse, genital irritation, familial stress, emotional deprivation and lack of breast feeding were found to have a positive correlation with childhood gratification behaviour [10,15-17]. No relationship was obtained between masturbation and factors such as age, gender, parental education, being a single-child, being a first born and the baby’s sleeping place in the study conducted by Tashakori A et al., [9]. The present study was on a small sample and it was not a case-control study to make any assumption on aetiological perspectives.

Childhood gratification can be diagnosed clinically based on meticulous history taking, careful evaluation of videoclips in presence of parents and sometimes by direct observation of the act [5,11,14,19]. Unnecessary investigations should be avoided. Once diagnosis is established, possibility of sexual abuse and sources of genital irritation have to be ruled out. Parents should be counselled and reassured about the benign nature of the entity. They should be taught that punishing the child or shouting at them does not help, rather attempt should be made to distract them during episodes and to engage them into another playful activity [19-21]. Some children, however, might require psychiatric evaluation and behavioural therapy.

Most of the data available on childhood gratification are derived either from case reports or from studies carried out on very small sample sizes. This holds true for the present study also.

Further prospective studies including cases and controls need to be carried out on sufficiently larger population size to understand why some children are more prone to these kind of behavious than others; and these children should be prospectively followed-up well into their adulthood to observe whether these early childhood behaviours impart any adverse effect on their adult psycho-sexual tendencies.

Gratification behaviour can be seen in young children, and it is not pathological. Owing to various religious and cultural taboos attached to this, it can be a cause of enormous anxiety and guilty feelings among parents and family members. All paediatricians should be aware of the condition and its various display patterns in order to diagnose them timely and to avoid unnecessary investigations and harmful therapy. Since paediatricians occupy a privileged place in all societies, they can play an important role to educate children and parents about normal sexual development and to help prevent or rectify misconceptions about masturbation or gratification habits.

Author’s contribution: BB- Concept, manuscript writing and critical revision; MK, AS- Manuscript editing; MD, SM, RK- Compilation of records, review of literature.