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From the Department of Child Neurology, Children’s Hospital of Pittsburgh, Pittsburgh, Pennsylvania; Children’s Hospital Boston, Boston, Massachusetts; Children’s Memorial Hospital, Epilepsy Center, Chicago, Illinois; and Department of Neurology, University of Rochester Medical Center, Rochester, New York
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ABSTRACT |
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 TOP ABSTRACT REPRESENTATIVE CASE REPORTSRESULTSDISCUSSIONREFERENCES |
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Methods. Twelve patients presenting to a pediatric movement disorders clinic with a suspected movement disorder were determined to have postures and movements associated with masturbation. We reviewed the clinical history, examination, and home videotapes of these patients.
Results. Our patients had several features in common: (1) onset after the age of 3 months and before 3 years; (2) stereotyped episodes of variable duration; (3) vocalizations with quiet grunting; (4) facial flushing with diaphoresis; (5) pressure on the perineum with characteristic posturing of the lower extremities; (6) no alteration of consciousness; (7) cessation with distraction; (8) normal examination; and (9) normal laboratory studies.
Conclusions. The identification of these common features by primary care providers should assist in making this diagnosis and eliminate the need for extensive, unnecessary testing. Direct observation of the events is crucial, and the video camera is a useful tool that may help in the identification of masturbatory behavior.
Key Words: movement disorders • neurology • masturbation • sexual behavior
Abbreviations: EEG, electroencephalography • PDC, paroxysmal dystonic choreoathetosis
The concept of childhood masturbation was recognized as early as 1909 by Still.1 Masturbation involves stimulation of the genitals and typically begins at 
2 months of age, although in utero masturbatory behavior has been reported also.2,3 Incidence of this behavior typically peaks at 4 years of age and again in adolescence, occurring in 90% to 94% of males and 50% to 60% of females at some point in their lives.2 Although a normal behavior in childhood, it often is unrecognized by families and caregivers, especially because genital manipulation frequently is absent.2,4,5 In addition, in young children, unusual postures and movements can occur during masturbation and may lead the primary care provider to infer that seizures, abdominal pain, colic, or other neurologic or medical problems are present.6–9 The literature is sparse in delineating common characteristics of masturbatory behavior, with isolated case reports describing the events, examination, and history. In almost all cases, extensive work-up has been performed, including magnetic resonance imaging (MRI), electroencephalography (EEG), intravenous pyelography, small bowel biopsy, and gastrointestinal barium swallow.8–14 In some cases, treatment with antiepileptic agents was initiated before establishing a diagnosis of masturbatory behavior.8,13,14 However, recognition of masturbatory behavior as an entity can eliminate the need for unnecessary tests. We report the clinical characteristics of masturbatory behavior in 12 patients who were referred for paroxysmal dystonic events. The purpose of this case series is to delineate the typical features of masturbatory behavior so that physicians can better recognize this disorder, prevent costly and invasive evaluations, and provide appropriate guidance to parents.
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REPRESENTATIVE CASE REPORTS |
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TOPABSTRACTREPRESENTATIVE CASE REPORTSRESULTSDISCUSSIONREFERENCES |
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Case 2
Case 2 involves a 3-year-old girl who was the product of a pregnancy complicated by transverse presentation resulting in a cesarean section. The neonatal course was unremarkable. Her mother first noted spells at 3 months of age. She began twisting her neck and upper trunk with associated "moaning." The spells lasted up to 1 hour but were not associated with any change in responsiveness or interaction. At 15 months, the movements evolved to include twisting of her trunk and arms. The episodes occurred up to 10 times a day but were more frequent when she was tired. Family history was significant for a sister with quadriplegic cerebral palsy and an older sister with aortic stenosis. Work-up and evaluation included an MRI of the brain and EEG, both of which were normal. Before referral, she was treated with trihexyphenidyl, baclofen, clonidine, amitriptyline, carbidopa/levodopa, and carbamazepine, all without change in the frequency or character of the episodes. She was referred to our movement disorder center, at which she was found to have mild global developmental delay, but she had normal general pediatric and neurologic examinations.
A home video showed her lying on her right side, with both hips flexed and adducted, the right knee was extended and the left knee was flexed, the ankles were plantar-flexed. Rhythmic pelvic rocking was present throughout the episode. The left arm was flexed at the elbow and wrist with the hand placed over her abdomen. The right arm was extended at the elbow and wrist. The neck was extended as well. Grunts, cries, snorting sounds, and whimpers were heard at times. The episodes tended to occur while she was getting ready to sleep. Her parents were consistently able to distract her during the episode, which caused the movements to pause or cease.
Case 7
Case 7 involves a 3-year-old girl who was the product of a pregnancy complicated by preterm labor treated with magnesium. Subsequent term delivery and postnatal course were unremarkable. At 5 months, she was noted to have episodes in which she would "tense up her body" and turn red. She was reported to be responsive throughout the episodes. The spells increased in frequency as she became older, occurring multiple times a day. She reported that she "had to stiffen." After longer episodes, she would respond affirmatively if asked whether her body "hurts." The movements were most likely to occur when she watched television, listened to stories being read to her, or was sleepy. Before referral she had an extensive work-up that included brain MRI, head computed tomography, EEG, and upper and lower gastrointestinal series, all of which were normal. She was treated with carbidopa/levodopa, carbamazepine, and several other antiepileptic medications, all without improvement. She was referred to our movement disorder center, at which she had normal general pediatric and neurologic examinations.
A home video showed her lying on her left side, partially prone, flexed at the hip and knees and plantar-flexed at the ankles. The left calf rested on top of the right knee or thigh. The left arm was extended and placed on the abdomen. She rhythmically contracted and relaxed the thigh muscles and tilted her pelvis. Occasional grunting and breath-holding were observed. At times, her face reddened and she displayed various facial expressions, grimaces, smiles, or staring with an open mouth. She consistently was able to stop the movements to talk or pay attention to a story being read to her.
Case 9
Case 9 involves a 4-year-old girl who began to have episodes of stiffening at 5 months of age (Figs 1 and 2; see also Videos 1 and 2, which is published as supporting information on www.pediatrics.org/content/full/VolNo/IssueNo/FirstPageNo). The episodes occurred when lying on her back and consisted of crossing and stiffening the legs, straightening the arms, making "grabbing motions" with the hands, and pursing the lips. During these episodes, her breathing became irregular and she perspired. They occurred multiple times a day, lasting from seconds to hours, but her parents could talk to her and stop the spells. She often told her parents that she couldn’t stop but "had to stretch." Her work-up included 2 brain MRIs, 2 prolonged EEGs, an upper gastrointestinal series, lumbar puncture, electromyography, and extensive laboratory testing, all of which were normal. She was described by her parents as being bright and energetic. Her development was normal, as were her general pediatric and neurology examinations.
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On subsequent follow-up at the age of 11 years, the parents reported that the patient continued to have occasional episodes of "stretching," precipitated by fatigue. She also has episodes of rocking backing back and forth, with thrusting movements of buttocks, while riding in a car.
Case 11
Case 11 involves a 2-year-old girl who was referred by a pediatric neurologist for spells of back-arching. She was a term infant born without complications. Approximately 1 year before evaluation, she developed spells that always occurred while sitting in a high chair. The episodes were described as starting with rhythmic movements of the right fingers, then rhythmic movements of the right arm, followed by "jerking" of all 4 extremities with back-arching, head tilt, and mouth-grimacing. She appeared responsive during each of the episodes. Her parents report that she responded to her name but appeared to have a "dazed," distant facial expression. Before referral, she had multiple studies including a routine EEG, MRI of the head, and laboratory testing for thyroid function, heavy metals, ceruloplasmin, antistreptolysin O titer, basic metabolic panel, lactate, and pyruvate. All studies were normal. Initially, based on the history, she was thought to have seizures. She had a trial of carbamazepine without any change in the episodes. She was referred for additional evaluation of these episodes. Her development was normal, as were her pediatric and neurologic examinations.
A home video showed several episodes occurring while she was sitting in a high chair. There were rhythmic, repetitive flexion and extension of the right fingers, sustained plantar flexion, and rhythmic pelvic thrusting against the seat of her high chair. During the episode, she appeared dazed, with grimacing and repetitive eye blinks. Occasionally, she demonstrated tonic extension of the trunk. The episodes had abrupt starts and stops. She was awake and responsive throughout each spell.
Case 12
Case 12 involves a 2-year-old girl who was referred by a pediatric neurologist for spells of posturing. She was a 32-weeks’-gestation infant born to a mother with mild mental retardation and drug and alcohol use during the pregnancy. The patient had no postnatal problems but was in foster care until 2 months of age, when she was adopted by her current adoptive parents. At 1 year of age, she developed posturing, usually while in a high chair or car seat. "Build-up" was noted in which she would tense and then sustain a twisting posture of both legs and occasionally of the hands. During these spells she would grunt and had periods of heavy breathing without loss of consciousness. She was able to respond to her parents throughout. They were able to distract her out of the episode, but it would require a strong motivating stimulus to get her to stop. Occasionally, she would continue to have "build-up" with tensing of the legs, then finally sigh and fall asleep. She was able to get up immediately and run around normally. Before referral, she had extensive testing because of these episodes, including several EEGs and brain MRIs, all of which were normal. A pH probe was normal. Genetic testing for the DYT1 mutation and the dentatorubropallidoluysian (DRPLA) triplet repeat was negative. She had medication trials with carbamazepine, phenytoin, lorazepam, and gabapentin, which did not change the frequency or the character of these events. She developed a rash while on carbamazepine. Her development was normal, although the parents thought that she was clumsier than other children her age. Pediatric and neurologic examinations were normal.
Video review showed several episodes occurring when she was sitting in a high chair or car seat. Occasionally the events occurred when awaking from a nap. In the event, 1 leg was crossed over the other, with plantar flexion bilaterally. Her thighs adducted and abducted in a rhythmic manner and was associated with pelvic thrusting. She also had rhythmic extension and flexion of her fingers with variable internal-external rotation at the shoulders. Facial flushing, facial grimacing, and diaphoresis accompanied these events. She appeared dazed and glassy eyed during the episodes. After 1 episode, she appeared to suddenly relax and fall asleep. During these spells, the parents were able to get her attention, with brief cessation of this rhythmic activity.
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RESULTS |
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TOPABSTRACTREPRESENTATIVE CASE REPORTSRESULTSDISCUSSIONREFERENCES |
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DISCUSSION |
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TOPABSTRACTREPRESENTATIVE CASE REPORTSRESULTSDISCUSSIONREFERENCES |
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In our case series, masturbation was previously misdiagnosed as a movement disorder, usually dystonia, resulting in extensive diagnostic testing in the majority of our patients and initiation of multiple medications. In his article on masturbatory behavior, Fleisher and Morrison9 described 12 features of the history, description of the events, and examination common to the clinical presentation of his 5 patients. We found similar common features in our patients that we feel distinguish masturbatory behavior. Onset occurred after the age of 3 months and before 3 years, with stereotyped episodes of variable duration. From all the children vocalizations with quiet grunting and irregular breathing could be heard. Facial flushing with diaphoresis often was present. Patients assumed characteristic posturing of the lower extremities with pressure on the perineum. Consciousness was preserved throughout the episodes, with cessation of the activity with distraction. In all cases, laboratory studies and examination were normal. The episodes usually occurred in the context of boredom or states of excitement or anxiety.
In our series, direct observation of these events was invaluable; this cannot be overemphasized. As a rule, in evaluating possible movement disorders, direct observation is essential for the clinician to make a diagnosis. The use of home videos for the assessment of tics has been described by Goetz et al.17 Because events can be fleeting, as well as environment and state dependent, they may not be witnessed in a limited office setting. Goetz et al found that when 3 environments were compared (home with family, home alone, and office), tics were more pronounced when the patient was home alone and not in the office. The severity and character of the tics were increased dramatically in some cases as compared with the office setting. Similarly, with masturbatory behavior, these events may not be witnessed in the office. What can be portrayed in the history as dystonia or seizures can appear differently on direct observation. Others have emphasized the importance of direct observation in masturbatory behavior as well.10,13 In case 12, a neurologist concluded that the patient had seizures by history, but when multiple medications failed, the conclusion was made that the symptoms represented dystonia. Direct observation was the key to diagnosis in this case.
Mistaken diagnosis by event description only is understandable. The spectrum of paroxysmal events in children, especially in the infant and toddler period, is broad.6,7 The distinction between masturbatory behavior and seizures can be difficult. Children with masturbatory behavior can appear to have altered consciousness with a glassy-eyed, fixed gaze. However, a key feature to masturbatory behavior is that it is volitional and can cease with distraction. Children will often appear annoyed if they are interrupted. In addition, torsional postures and rocking are uncommon in seizures.8,13 Another diagnostic consideration is paroxysmal dystonic choreoathetosis (PDC). Episodes of PDC can occur bilaterally or unilaterally without triggering factors and last for variable periods of time.18,19 Children under the age of 1 year can be affected. In contrast to PDC, however, children with masturbatory behavior can abruptly and easily move out of their assumed postures. However, even for experienced movement disorders specialists, the distinction between paroxysmal movement disorders and masturbatory behavior can be difficult.20
Once the diagnosis of masturbatory behavior is made, a history of sexual abuse and perineal irritation should be ruled out.2,9 Masturbatory behavior has been reported in sexually abused children.21 Perineal discomfort such as that caused by vulvovaginitis, urinary tract infections, or dermatitis may exacerbate the behavior but may also be the result of the behavior.13,16 The exact relationship between perineal irritation and masturbatory behavior is unclear: perineal irritation may intensify the behavior and increase the frequency with which it is performed. On the other hand, daily application of friction to the perineum may precipitate irritation of the region. In either case, an external genital examination should be performed, and perineal irritation should be treated. If the physical and neurologic examinations are normal, no additional diagnostic testing needs to be performed. Reassurance for the family is the key to management, with redirection should the behavior prove embarrassing for the family or occur in public.2,9 The parents should be educated that this is a normal behavior resulting from random exploration of the body by the infant.2 Masturbation likely is initiated when the infant discovers that certain maneuvers can bring about a pleasant and comforting sensation. It may be viewed in the same category as thumb-sucking, body-rocking, or other behaviors that infants use to enhance comfort. This behavior may subside as the infant becomes engaged in other objects or activities in the environment. Again, the parents should be reassured that this is a normal behavior and it may be seen in infancy.7,10,14,16
Little is available in the literature regarding long-term follow-up of children with masturbatory behavior. In a study on normal sexual behavior in children, sexual behavior that appeared most frequently included self-stimulating behaviors and peaked at 5 years of age for both boys and girls, dropping off over the next 7 years.4 For the most part, these children have normal development and the behaviors do subside. Providers should note that masturbation is a behavior seen at all ages, as it was for the girl in case 9, who, at the age of 11 years, continues to exhibit this behavior, particularly in times of mild anxiety.
We suggest a few practical points for management of masturbatory behavior:
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FOOTNOTES |
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Address correspondence to Jonathan W. Mink, MD, PhD, Department of Neurology, University of Rochester Medical Center, 601 Elmwood Ave, Box 631, Rochester, NY 14642. E-mail: jonathan_mink{at}urmc.rochester.edu
No conflict of interest declared.
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