Nonsyndromic craniosynostosis associated with bipolar affective disorder and mild cognitive disorder

Dear Editor, Craniosynostosis is a prevalent congenital anomaly, impacting around one in 2000 to 2500 live births, and is defined as the premature fusion of one or more cranial sutures which leads to deformed craniofacial appearance (normal age of suture closure is 18 months to 2 years).[1] Craniosynostosis is broadly divided into two groups- syndromic craniosynostosis and nonsyndromic craniosynostosis. The former is associated with genetic factor, such as single-gene mutations, polygenic factors, or chromosomal structural abnormalities.[2] By contrast, nonsyndromic craniosynostosis, which constitutes almost 85% of all cases, is multifactorial and may have genetic and environmental influences. It can be categorized based on the involved cranial sutures into sagittal, coronal, metopic, lambdoid, and complex types.[3] Of these, sagittal craniosynostosis stands out as the most prevalent type, with an incidence of around 1 in 5,000 births, constituting 40–55% of all nonsyndromic cases. Coronal synostosis is the second most common followed by metopic, lambdoid, and complex types in that order.[4] A young female who presented to us with symptoms of bipolar affective disorder and cognitive decline with abnormal skull shape alienation is described.[5] A 23-year-old female, undergraduate student, with history of premature birth at 07 months, without any family history of psychiatric illness, with previously normal level of adaptive and cognitive functioning presented in April 2022 with insidious onset and gradually progressive irritability on trivial matters interspersed with undue cheerfulness. She was overtalkative, had decreased need for sleep, overenthusiastic for “Swatch Bharat Abhiyan,” and would tirelessly clean garbage on roadside. She was started on some medications by a psychiatrist (documents not available), and her symptoms improved over two months. Subsequently, in August 2022, she reported experiencing low mood, social withdrawal, and a diminished interest in pleasurable activities, easy fatiguability, initial insomnia, poor scholastic performance, and headache (holocranial) for 20 days. Physical examination revealed BMI of 28 kg/m2 and abnormal head shape (scaphocephaly) with a head circumference measuring 60 cm. Strabismus, Rt eye was present. Mental status examination revealed kempt, cooperative individual with decreased psychomotor activity, increased speech latency, decreased rate, and volume, sad mood as sad and depressed effect with reduced range and reactivity. There were no hallucinations. Her score on the Beck depression inventory was 20, minimental state examination (MMSE) 23, and Montreal cognitive assessment (MoCA) 24. On the PGI battery of brain dysfunction (PGI-BBD), she had dysfunction rating of 12 in PGI memory and verbal adult intelligence scale. Hematological and biochemical parameters were within normal limits. Her MRI brain was suggestive of craniosynostosis with premature fusion of sagittal suture (scaphocephaly). With a diagnosis of bipolar affective disorder, current episode moderate depression, without somatic syndrome, she was started on tab bupropion 150mg OD and tab divalproex sodium 500 mg BD. Neurosurgeon diagnosed her with nonsyndromic craniosynostosis and prescribed tab topiramate 50mg/day for headaches. The patient showed improvement from the 2nd week onward achieved remission within the next one month and functionality in the social sphere. However, cognitive decline continues to persist. Existing studies with limited data indicate that children diagnosed with nonsyndromic craniosynostosis are more prone to receiving diagnoses of psychiatric disorders when compared with unaffected children in the general population.[5] Frequent psychiatric disorders encompass intellectual disability, language disorders, neurodevelopmental conditions such as autism spectrum disorder, attention-deficit hyperactivity disorder, various behavioral and emotional disorders, cognitive decline, and specific learning disorder.[6] Other psychiatric disorders such as psychotic disorders (behavioral abnormalities), bipolar affective disorder, depressive disorder, eating disorder, anxiety disorder, personality disorder, and suicide attempts may also be present in nonsyndromic craniosynostosis.[7] Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.

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