Enteric fever presenting as catatonia syndrome – Case report

Case report

An 18-year-old male, without any identified comorbidity, presented on 17 December 2020 by his parents observing him to be aloof for the previous two days. They also noticed him to be less involved in his daily routine, being confined to his bed and to have decreased speech. On further questioning, a history of febrile illness, documented by a high-grade fever with chills, frontal head ache and a few episodes of vomiting from 2–11 December 2020, was identified. He had already taken oral ciprofloxacin 1 g/day for three days prior. He was not on any other long-term medication and denied any recreational substance use.

Clinical examination revealed an averagely built and moderately nourished male, who was afebrile with normal haemodynamic parameters except for a relative bradycardia of 62 beats/min with regular rhythm. He was conscious, drowsy and aloof. After repeated questioning, he was found to have insight and was oriented to time, place and person. He had a coated tongue, expressionless facial features, posturing (holding any posture kept by the physician against gravity) with a halting, slow stiff gait. His systemic examination was unremarkable except for mild non-tender hepatomegaly, extending 2 cm below the right subcostal margin in the mid-clavicular line. He had no focal neurological deficit on admission.

A differential diagnosis of cerebral malaria, typhoid encephalopathy, septic or metabolic encephalopathy were considered. Laboratory parameters revealed a normal leucocyte count (5.7 × 109/m), high C – reactive protein level (190.3 mg/L) and thrombocytopenia (73 × 109/m). Liver enzymes were mildly raised (AST – 165 U/L, ALT – 92 U/L, bilirubin – 5.13 µmol/L, ALP – 103 U/L); electrolytes, peripheral blood picture and kidney function tests were within normal ranges. Serum procalcitonin, a rapid malarial antigen test, dengue and scrub typhus serology (IgM) and COVID 19 RT–PCR were negative. However, Typhi-dot IgM was positive in high titres, and a salmonella blood culture was sought. A chest radiograph and an urgent contrast-enhanced MRI brain scan and CSF examination were all normal.

He was started empirically on intra-venous ceftriaxone 2 g/day, gentamicin 80 mg 8 hourly and oral quinine while the investigations were pending.

A specialist psychiatrist diagnosed catatonia (based on the Bush–Francis Catatonia Rating scale score of 13/42) secondary to organic disease and started oral lorazepam 4 g/day in divided doses. A Widal test revealed a Salmonella typhi ‘O’ titre of 1:320 and blood culture grew S. typhi which was sensitive to ceftriaxone and other agents. Quinine was withdrawn at this point.

Catatonic symptoms progressively improved after three days, and he was discharged after seven days. Follow-up review after two weeks revealed no psychiatric features, and the abnormal lab parameters (thrombocytopenia and hepatic enzymes) had returned to baseline.

Discussion

Neuro-psychiatric manifestations in the natural disease course of enteric fever are not uncommon, though isolated catatonia is unusual in clinical practice, particularly during defervescence.

Neuro-psychiatric symptoms were observed in studies by Osuntokun et al. ¹ and Ali et al. ² in 57 and 84% of enteric fever patients, respectively. In a community-based typhoid outbreak of 2009 in Malawi, ³ they were found in 13%. Numerous isolated case reports and series have reported from mild confusion to typhoid encephalopathy, although isolated catatonia syndrome is a rare association.^4–10 Our patient did not have co-existing syndromic depression, psychosis or delirium as commonly reported.^1–3

Multi-factorial causation is highly likely including metabolic derangements, toxins and auto-immune phenomenon, though susceptible personality traits have been a proven risk factor. ¹¹ However, detailed serological and autopsy studies of such patients have failed to provide any valid evidence to support any of the above causes and so remain ambiguous.^1,4,12 A proposed mechanism of neurotransmitter imbalance by Jain et al., ¹³ especially the dopaminergic–cholinergic system, remains plausible in our scenario. Although the prognosis is usually favourable, prompt identification of any neuro-psychiatric symptoms in enteric fever needs to be taken seriously.