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CASE REPORT
Year : 2009  |  Volume : 31  |  Issue : 2  |  Page : 92-95 Table of Contents   

A case of corpus callosum agenesis presenting with recurrent brief depression


1 Department of Psychiatry, Calcutta National Medical College, Kolkata, India
2 Department of Pediatrics, Nil Ratan Sirkar Medical College, Kolkata, India

Date of Web Publication21-May-2010

Correspondence Address:
Ranjan Bhattacharyya
29, Anandasree, Garia, Near Boral TB Hospital, Kolkata - 700 084
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0253-7176.63580

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   Abstract 

Agenesis of corpus callosum can have various neuropsychiatric manifestations. Following case report highlights the case of a young man presenting with features of recurrent brief depressive disorder, each lasting for about 3 to 7 days, for over a year. He had history of occasional headache and episodes of swooning attack in between, usually precipitated by emotional events. His neuroimaging revealed agenesis of corpus callosum. He was experiencing swooning attacks as he became aware that some 'unusual' findings were present in his reports. Recurrent brief depression can be a manifestation of this congenital anomaly, and conversion disorder can be present as comorbid diagnosis perhaps due to ignorance and fear of this apparently innocuous congenital malformation.

Keywords: Conversion disorder, corpus callosum agenesis, recurrent depression


How to cite this article:
Bhattacharyya R, Sanyal D, Chakraborty S, Bhattacharyya S. A case of corpus callosum agenesis presenting with recurrent brief depression. Indian J Psychol Med 2009;31:92-5

How to cite this URL:
Bhattacharyya R, Sanyal D, Chakraborty S, Bhattacharyya S. A case of corpus callosum agenesis presenting with recurrent brief depression. Indian J Psychol Med [serial online] 2009 [cited 2019 Nov 18];31:92-5. Available from: http://www.ijpm.info/text.asp?2009/31/2/92/63580


   Introduction Top


Agenesis of corpus callosum (ACC) is mostly a congenital anomaly that occurs predominantly in males, either isolated or in combination with other CNS or systemic malformations. Corpus callosum develops from the lamina reuniens in the telencephalon and begins to appear at around 10.5 weeks, till it reaches adult form by 17 weeks. The occurrence of ACC is usually sporadic. Disturbance of embryogenesis in the first trimester of pregnancy by an unknown insult is sometimes put forward as a probable antenatal cause. Once thought to be a rare condition, ACC has been turning up with increasing frequency ever since the widespread clinical use of MRI. ACC can occur in other congenital anomalies, including  Arnold- Chiari malformation More Details More Details, Dandy-Walker syndrome, schizencephaly (clefts in brain tissue) and holoprosencephaly (failure of the forebrain to divide into lobes), Aicardi's syndrome, fetal alcohol syndrome and several trisomies. [1] ACC can also be associated with malformations in other parts of the body, such as midline facial defects, or can occur as one of the developmental abnormalities in schizophrenia. The effects of the disorder range from subtle to severe, depending on associated brain abnormalities. Intelligence may be normal with mild compromise of skills requiring matching of visual patterns. But children with the most severe brain malformations may have intellectual disability, seizures, hydrocephalus and spasticity. [2],[3]

Previous literatures have mentioned about various neuropsychiatric presentations of this congenital anomaly. Depressive spectrum disorders have been mentioned in various literatures to be present with this anomaly, but recurrent brief depression has not been mentioned in any of them. [3,4] This congenital anomaly has also been found in bipolar disorder, [5] schizophrenia [6] and can interfere with psychomotor development. [7] In addition, individuals with ACC are reported to have more sensory deficits and abnormalities, as well as altered patterns of feeding/eating, elimination and sleep. [8]


   Case Report Top


A 25-year-old man residing in a suburban region was brought to the Psychiatry OPD at the tertiary care hospital with biparietal dull-aching headache lasting for about an hour, occurring 3 to 4 times per month for the past 2 years. The headache, to start with, was spontaneous in onset, without nausea or vomiting, diurnal variation, fever, photophobia, phonophobia, and subsided with analgesics tablet ibuprofen 400 mg only. He had been prescribed tab Flunarizine 10 mg OD and Amitriptyline 25 mg OD for the last 1 year. On careful history-taking from the informant (father) and the patient himself, it was known that the patient had suffered from episodes of depressed mood and irritability every month lasting for 3 to 7 days on an average, diminished interest in pleasurable activities, hypersomnia, psychomotor retardation, fatigue and loss of energy for the past 1 year with recurrent thoughts of death but without any history of suicidal attempt so far. There was no positive family history of any major medical or psychiatric illnesses in the family up to three generations. He had been advised to have a CT scan followed by an MRI of brain for the persistent headache, 1 month back. Also there was a history of palpitation, breathlessness, clenching of teeth followed by unresponsiveness, each episode lasting for 30 minutes to 1 hour, four times in the past 2 weeks. There had been no tongue bite, incontinence, loss of consciousness during the episodes, and the patient was occasionally verbalizing during these periods. He had been brought to the emergency room 1 week back by his family members and neighbors in a state of unresponsiveness. The reported symptoms had lasted for 2 hours, along with jerky movements of all four limbs and clenching of teeth but without any injury, incontinence or fall. According to his parents, his problems started suddenly after he came to know his MRI brain findings. He was able to adjust his body parts when placed in awkward position during these periods of unresponsiveness. After gaining consciousness, he refused to talk or answer but tried to use gestures and sign languages to answer questions asked to him. His vocalization was normal as tested by cough reflex. He had a fall when he was asked to walk a few steps, but the fall was guarded without any injury.

During mental status examination (MSE), the patient was found to be guarded initially, with increased reaction time, monotonous speech and depressed affect. Neuropsychological testing revealed subtle differences in higher cortical function. He suddenly had an attack mimicking a swooning attack in the presence of his family members while the interview was in progress, which subsided on negative reinforcements and suggestions. There were no clinical features suggestive of an epileptic seizure, which was confirmed by routine EEG and subsequently by video-EEG.


   Discussion Top


Agenesis of corpus callosum is a rare condition (with US frequency rates reported to be 0.07% to 5.3%) which usually presents with clinical features such as seizures, feeding problems, developmental delay, impaired hand-eye coordination, impaired visual and auditory memory and hydrocephalus. In this patient, there were few delayed milestones of development only without any gross physical or mental disability. (IQ was 77 as per WAIS-IV version.) No other congenital anomalies were found. The uniqueness of the case was that it presented with recurrent depressive disorder with swooning attacks. The patient responded well to a trial of benzodiazepines (Clonazepam) followed by maintenance with SSRIs. The diagnosis has been done on the basis of appendix B of the Diagnostic and Statistical Manual of Mental Disorders (DSM-IV-TR), which includes criteria and axes provided for further study. The diagnosis remained stable on subsequent follow-up visits and did not turn out to be any other major mood disorders. Though the condition can have wide range of manifestations, in our case we noticed only recurrent brief depression de novo and conversion disorder as a consequence of reaction when the diagnosis was disclosed to the patient. He never met the criteria of major depressive disorder during the entire period of his illness.


   Conclusions Top


The clinical manifestations of callosal agenesis can be described under two headings, viz., nonsyndromic and syndromic. Nonsyndromic forms are the most common. An unknown, though probably small, proportion of patients are completely asymptomatic, or more commonly, their condition is incidentally discovered during neuroimaging. [9] The recurrent brief depression is an entity mentioned in ICD-10 (International Classification of Mental and Behavioral Disorders) in the section of other recurrent mood (affective) disorders (F 38.10). The DSM-IV-TR demands further study to formulate a more valid diagnosis of this entity. The questions of stability and validity of its diagnosis are always there since its nosology. In this case, it has been diagnosed with another rare congenital CNS anomaly, the agenesis of corpus callosum (ACC). Psychiatric antecedents and comorbidities in agenesis of corpus callosum (ACC) need to be explored more in future case reports. Similarly comorbid general medical conditions need to be studied for getting more information on the diagnosis of recurrent brief depressive disorder. [ 10][Figure 1],[Figure 2],[Figure 3],[Figure 4],[Figure 5]

 
   References Top

1.Barkovich AJ, Norman D. Anomalies of the corpus callosum: Correlation with further anomalies of the brain. AJNR Am J Neuroradiol 1988;9:493-501.   Back to cited text no. 1      
2.Jeret JS, Serur D, Wisniewski KE. Clinicopathological findings associated with agenesis of the corpus callosum. Brain Dev 1987;9:255-64.   Back to cited text no. 2      
3.Badaruddin DH, Andrews GL, Bφlte S, Schilmoeller KJ, Schilmoeller G, Paul LK, et al. Social and behavioral problems of children with agenesis of the corpus callosum. Child Psychiatry Hum Dev 2007;38:287-302.  Back to cited text no. 3      
4.David AS, Wacharasindhu A, Lishman WA. Severe psychiatric disturbance and abnormalities of the corpus callosum: Review and case series. J Neurol Neurosurg Psychiatry 1993;56:85-93.  Back to cited text no. 4      
5.McCrea SM. Bipolar disorder and neurophysiologic mechanisms. Neuropsychiatr Dis Treat 2008;4:1129-53.  Back to cited text no. 5      
6.McGrath JJ, Richards LJ. Why schizophrenia epidemiology needs neurobiology-and vice versa. Schizophr Bull 2009;35:577-81.  Back to cited text no. 6      
7.Galdσs M, Martνnez R, Prats JM. Clinical outcome of distinct Aicardi syndrome phenotypes. Arch Soc Esp Oftalmol 2008;83:29-36.  Back to cited text no. 7      
8.Doherty D, Tu S, Schilmoeller K, Schilmoeller G. Health-related issues in individuals with agenesis of the corpus callosum. Child Care Health Dev 2006;32:333-42.  Back to cited text no. 8      
9.Bossy JG. Morphological study of a case of complete, isolated, and asymptomatic agenesis of the corpus callosum. Arch Anat Histol Embryol 1970;53:289-340.  Back to cited text no. 9      
10.Lφvdahl H, Andersson S, Hynnekleiv T, Malt UF. The phenomenology of recurrent brief depression with and without hypomanic features. J Affect Disord 2009;112:151-64.  Back to cited text no. 10      


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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