Partial Agenesis of Corpus Callosum And Its Neuropsychiatric Manifestations: Case Report And Review Of Literature

Case Reports in Psychiatry
05 Apr, 2019 ,

Case of a 45-year-old patient with partial agenesis of the corpus callosum who exhibits depressed mood, transient loss of memory, and history of cognitive, social, and behavioral disturbances that developed during his childhood. Recent and pertinent literature has been reviewed and the agenesis of the corpus callosum and its associated neuropsychiatric manifestations are discussed.

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We describe the case of a 45-year-old Hispanic man who presented to the psychiatric emergency room on account of depressed mood and forgetfulness. He was found by his niece sitting in the bathroom batting away imaginary flies and crying, stating that he could not remember anything which prompted his niece to call emergency medical services (EMS).

The patient was emotionally labile and could not remember his name or address at the time of presentation. He was hyperverbal and difficult to interrupt, and his speech was disorganized. The patient stated that prior to admission, he left his home and suddenly could not remember how he got to the location he had traveled to. He then returned home and entered the bathroom to look for a belt to hang himself with, because he could not remember any of the evening’s events. He stated that he felt lonely and helpless and that he had suicidal thoughts. The patient stated that his sleep had been poor. He endorsed a perceptual disturbance of seeing fleas that were trying to infest his body. He also endorsed an auditory hallucination of a male voice calling his name. Collateral information from his niece, who called the EMS, revealed that the patient had been acting bizarre with two previous episodes of new-onset wandering behavior in the past six months, both associated with heavy alcohol use. She also reported that the patient had a 15-year history of schizophrenia and that he had had similar episodes in the past, which were usually brief and resolved without the need for hospitalization. During a similar episode three years ago, the patient began attacking his family members and was hospitalized after the police were called. The patient also received a diagnosis of major depressive disorder five years ago. The patient was admitted to the inpatient psychiatry unit with a diagnosis of major depressive disorder. Urine toxicology at the time of admission was negative for controlled substances, illicit drugs, and alcohol. The patient’s admission Complete Blood Count (CBC) and kidney liver function tests were within normal limits. Rapid regain test was negative. Serum sodium and potassium were 138mmol/L (136–144.0mmol/L) and 4.4mmol/L (3.6–5.1), respectively. Other routine urine analyses and coagulation profiles were also within normal limits as were routine chest radiograph and ECG. Serum thyroid stimulating hormone was below the lower limit of normal 0.409 uIU/ml (0.450–4,500 uIU/ml) and free T4 was 1.09 ng/ml (0.82–1.77 ng/dL). The patient had no symptoms of hyperthyroidism.

Other chemical laboratory investigations were within normal limits except for dyslipidemia. Computerized tomography (CT) and magnetic resonance imaging (MRI) performed during admission revealed partial agenesis of the corpus callosum with the absence of the posterior body and the splenium.

On day 1 of hospitalization, the patient was hyperactive and restless on the unit. He was treated with escitalopram 10 mg PO daily and risperidone 2 mg PO BID. By day 2 of hospitalization, the patient was able to recall his name and his perceptual disturbances resolved, but he was still hyperverbal, with increased activity. By day 9 of hospitalization, the patient’s condition had stabilized, and he was discharged.

According to the patient’s mother, the pregnancy was reported to be complicated at five months, and the patient was born at seven months. He had normal gross motor development, but language was delayed until the age of 7 years. The mother reported a history of cognitive developmental delay and intermittent behavioral disturbances that led to his dropping out of school in fifth grade.

Literature Review

We conducted a literature review to explore various neuropsychiatric manifestations of agenesis of the corpus callosum presented in this case report. This was focused on identifying peer-reviewed articles related to partial agenesis of the corpus callosum and pathology. We searched PubMed, EBSCO, and Web of Science for articles on the function of the corpus callosum and presentation when absent or when there is a lesion without language restriction or restriction to time-period. The search was conducted using the keywords and MeSH terms: “function” “agenesis” “corpus callosum”, “schizophrenia”, and “psychosis.” Screening for eligible articles was conducted independently by six authors. Eligible studies were those that focused on the symptomatology of agenesis of the corpus callosum or defect in the corpus callosum and the respective psychiatric manifestations in human subjects. The citation manager used was Endnote in order to prevent duplication of references.

Because of the rarity of agenesis of the corpus callosum, all types of studies were considered for analysis including experimental, meta-analysis, cohort, case-control, case series, and case reports. The pertinent information from the included articles was abstracted and entered into a data abstraction form using Microsoft Excel constructed by the group. The information extracted from eligible articles includes the title of the article, gender and age of the patient, signs, symptoms elicited, and lab findings/imaging recorded.

All the reviewed studies reported the gender of patients with 56 percent being male and 44 percent female. The ages of the subjects ranged from 3 to 73 years; the mean age was 33 years. Furthermore, out of the 15 patient in the reviewed case reports and case series identified to have either a partial or complete agenesis of the corpus callosum about 36 percent were female and 64 percent were male.

Reported symptoms ranged from changes to behavior and personality, including paranoia, hallucinations, and delusions, to cognitive impairment. Radiological findings of the patients varied; however it mainly included agenesis or atrophy of the corpus callosum, followed by lipoma of the corpus callosum.