ABSTRACT
Attention-deficit/hyperactivity disorder (ADHD) is a disorder characterized by inattention, hyperactivity, and impulsivity, causing social, academic, emotional, and cognitive difficulties. ADHD symptoms start before age 12, and in some children, they’re noticeable as early as 4 years of age. This case documents an ADHD case accompanying agenesis of the corpus callosum (ACC) who showed clinically significant improvement after 12 occupational therapy sessions. Sensory integration therapy, which is a branch of occupational therapy, was applied to the patient by an occupational therapist for approximately 12 sessions. After 3 months of occupational therapy sessions, there was six-month progress on the Denver Development Test 2. The ACC, an organic pathology, may lead to academic, psychiatric, and social problems. Patients can benefit from pharmacological treatment, physical therapy, speech therapy, physiotherapy as well as occupational therapy. Treatment is symptomatic according to the patient's current problem, and early intervention and supportive treatment are critical. Most children need individual rehabilitation, even if their intelligence quotient remains within the normal range.
Introduction
Attention-deficit/hyperactivity disorder (ADHD) is characterized by inattention, hyperactivity, and impulsivity, a common neuropsychiatric disorder in which symptoms usually begin around the age of four years. This disorder can cause social, academic, emotional, and cognitive turmoil. The prevalence of ADHD in school-age children varies between 5.0 and 10.0%.1 Although the cause of ADHD has not been yet been clarified, the deterioration in the early developmental stage is caused by various genetic and environmental factors2. This article documents a case of ADHD accompanying corpus callosum agenesis showing clinically significant improvement after twelve occupational therapy sessions. The development of the Corpus Callosum begins with the formation of primitive lamina terminals in the sixth week of fetal life3. Structural formation continues between the 8th and 20th weeks of pregnancy. Meanwhile, radiation, infectious and chemical agents, chromosomal defects, maternal hormones, and nutritional disorders may cause complete or partial defects of the corpus callosum4. Agenesis of the corpus callosum (ACC) is a white matter anomaly characterized by the absence of a main interhemispheric commissure. The prevalence of ACC, which is the most common brain malformation, is 3-7/10005. The corpus callosum is a neuroanatomical region that connects stimuli from the cortex to the contralateral hemisphere, aiding sensory and cognitive processes. Communication and perception problems are observed in the anatomical problems of this structure. Although its characteristics in adults are well known, its mechanism(s) remain to be elucidated in the prenatal, early childhood, and childhood stages. A large body of literature examining the relationship between callosal dysgenesis/agenesis and clinical reflections has produced inconsistent findings. Most of the clinical findings in patients with ACC are mental retardation (60.0%), vision problems (33.0%), speech delay (29.0%), seizures (25.0%), abnormal muscle tone (25.0%), and nutritional problems (20.0%)6. If there is no developmental delay or mental problem, clinical symptoms such as mild behavioral or social problems, cognitive dysfunction, and ADHD may be seen7. Cognitive dysfunction includes learning difficulties, intellectual impairment, and mental retardation and ranges from mild (2/3 of cases) to moderate and severe (1/3 of cases). Behavioral or psychiatric disorders include ADHD, movement disorder, balance problems, speech disorders, learning difficulties, emotional disturbances, and autistic symptoms8. Despite these problems, functioning may be nearly intact.9,10
Case Report
A 4-year-old male patient was admitted to the outpatient clinic with complaints of sudden anger attacks, cognitive abnormalities, poor motor coordination, hyperactivity, restlessness, excessive talkativeness and difficulties in problem solving and planning. Physical examination findings were normal (no dysmorphic features, no hypotonia, ataxia, autistic behavior, seizures, or speech problems). On clinical examination, distractibility, hyperactivity, disinhibition, and inability to maintain attention were evident. In the developmental history of the patient, it was learned that milestones such as ambulation and speech were delayed. The teacher form stated that the patient was constantly on the move, had difficulty paying attention to the given tasks, and frequently quarreled with his friends. No features were identified in his family history. The Denver Developmental Screening Test 2 was applied to the patient. The Denver test revealed a 1-year delay in fine motor-adaptive and personal-social areas according to the age of the patient.
Routine blood tests and electrocardiogram were normal. Since neuro-genetic syndromes may also accompany hyperactivity, a neurology consultation was requested. While the electroencephalography was normal in neurological examination, magnetic resonance imaging showed asymmetry in the cerebral hemisphere, agenesis of the carpus callosum, and colpocephaly. The patient was started on 0.25 mg risperidone treatment for complaints of hyperactivity, restlessness, and excessive speech. During the follow-up, the dose was rearranged to 0.50 mg and the patient’s complaints were controlled. After obtaining informed consent from the child’s parents, occupational therapy sessions were started. Sensory integration therapy, which is a branch of occupational therapy, was applied to the patient once a week (approximately 12 sessions) for 3 months.
After the sensory integration therapy sessions, six-month progress was achieved in fine motor-adaptive and personal-social areas on the Denver Developmental Test 2. According to the renewed Denver Developmental Test 2, personal social development was determined as 3 years-6 months, gross motor development 4 years-3 months, fine motor-adaptive 3 years-6 months, and language 4 years-3 months. The final exam was better than before in perceiving instructions, cognitive functioning, and cooperating with others.
Discussion
Agenesis of the corpus callosum is one of the most common brain malformations in children with developmental delays. It is a heterogeneous condition (an isolated or congenital condition) and is more common in males than in females (2: 1)11. These children may present with mental retardation, vision problems, microcephaly, epilepsy, and behavioral, social, and attention-related symptoms. This case did not have epilepsy, but febrile seizures are more common in the ACC than in the normal population. Additionally, problems related to the corpus callosum may cause deterioration in areas such as cognitive, attention, language, learning skills, and impulse control mechanisms7. Because of these symptoms, the probability of being diagnosed with some psychiatric disease such as ADHD may increase. The impact of the structural change in the brain throughout the patient’s clinic was not fully understood in ADHD, but it was clear that there was a neurodevelopmental impairment. Moreover, microstructural changes in the corpus callosum were also seen in ADHD12. We could not be sure whether ADHD might be related to ACC in this patient, or whether these two conditions were independent of each other. More case-control studies are needed to understand the relationship between ADHD and ACC. Therefore, neuro-radiological tools are of great importance to exclude corpus callosum agenesis, especially in cases with developmental delay. Here, we focused on occupational therapy, which has a beneficial effect on the patient’s developmental delay, rather than the etiological underlying causes of this comorbidity. Additionally, we observed that sensory integration therapy improved the current clinical situation in this patient with developmental delay and hyperactivity complaints. Within the case of sensory integration therapy; the Wilbarger approach, deep pressure assessment, vestibular studies in a platform swing, proprioceptive applications in the ball pool, and selective visual stimulus therapy applications were applied to the patient13.
Occupational therapy, which increases the participation of patients in daily activities and to support areas where they have difficulties, is gaining increasing value today. When determining the interventions to be applied to the patient, the patient’s limitations and sensory and motor skills should also be taken into account13. Sensory integration interventions, especially applied to children with developmental delay, can be considered an early support program while focusing on increasing the patient’s participation in activity areas. Organizing an individual early support program is essential for children to reach their full potential.
Agenesis of corpus callosum is an organic pathology and may lead to academic, psychiatric, and social problems. Most children need an individual rehabilitation program, even if their IQ remains within normal limits. These patients may benefit from many treatments, including pharmacological and neurological treatment, auditory rehabilitation, physical therapy, occupational therapy, and speech therapies. Treatment is symptomatic, and early intervention and supportive treatment are critical for the child to reach the normal developmental stage
Ethics
Informed Consent: Informed consent was obtained from the parents.
Peer-review: Internally peer-reviewed.
Financial Disclosure: The author declared that this study received no financial support.
