Abstract
Keywords
Case report
A 32-year old male farmer was admitted with gait abnormality, altered mental status with agitation, decreased verbal output and responsiveness to commands of six days. These symptoms were preceded by low-grade fever for three days which subsided without any intervention. There was no associated history of headache, vomiting, seizure, focal neurological deficit, bowel and bladder incontinence nor loss of consciousness. Our patient was a known intravenous drug (opioid) user for three years and likewise a cigarette smoker. He had no other prior co-morbidity.
Initially, at a local hospital, he received single doses of lorazepam and haloperidol. He presented with tachycardia, a blood pressure of 140/96 mmHg, and oxygen saturation of 98% on room air. There was profuse sweating over his forehead suggesting autonomic dysfunction. His tone was symmetrically increased with cogwheel rigidity in all limbs; he had neck rigidity in its entire range of movement but normal muscle power.
Laboratory investigations revealed a leucocytosis (18.9 × 109/L), with neutrophilic (81%) predominance with raised urea and creatinine (15.15 mmol/L and 150.28 µmol/L, respectively). Further investigations showed aspartate aminotransferase of 1880 U/L and alanine aminotransferase of 1010 U/L.
In view of his febrile encephalopathy, ceftriaxone and acyclovir were administered. However, further investigations showed raised serum creatinine phosphokinase of 9040 U/L (range 26–308 U/L) along with lactate dehydrogenase (LDH) of 508 U/L (135–225 U/L). Urine myoglobin was also raised to 17,562,000 µg/L (Table 1).
Cerebrospinal fluid examination revealed total cell count of 18 with 91.5% lymphocytes and 8.5% neutrophils, protein 88 mg/dL and normal glucose with negative gram stain, culture, cryptococcal antigen and herpes simplex virus polymerase change reaction.
He no longer had a fever but an electro-encephalogram showed diffuse delta range slowing suggestive of diffuse encephalopathy. A brain CT scan showed diffuse cerebral oedema. But MRI scan did not show any findings suggestive of meningo-encephalitis.
The possibility of neuroleptic malignant syndrome due to haloperidol was entertained and bromocriptine 2.5 mg bd was administered, later increased to 5 mg tds together with lorazepam 1 mg tds. Improvement was seen over five days in his sensorium, rigidity, dysautonomia, and decrease in creatinine phosphokinase and serum LDH levels allowing discharge.
Discussion
Neuroleptic malignant syndrome (NMS) is a fatal condition linked to the use of anti-psychotic medications. Clinical manifestations include fever, altered sensorium, rigidity and autonomic dysfunction. NMS is diagnosed on the basis of clinical history, physical examinations findings and blood investigations. 1 NMS can have heterogeneous presentations.
Two core features of severe muscle rigidity and fever should be present after recent start or modification in dosage of an anti-psychotic medication, together with two or more of the following symptoms: diaphoresis, dysphagia, tremor, incontinence, changes in level of consciousness, mutism, tachycardia, elevated or labile blood pressure, leucocytosis, and elevated creatinine phosphokinase levels. 2
These clinical manifestations should not be attributable to another substance, neurological or medical condition. The risk of developing NMS has not been shown to be linked to dose or drug duration. 3
Typical clinical manifestations start with muscle rigidity followed by fever within a few hours, leading to altered mental status ranging from mild drowsiness, agitation, or confusion to a severe delirium or coma. In a study of 340 cases, 70% patients followed a usual course of mental status changes followed by rigidity, then hyperthermia and autonomic dysfunction. 4
There may be significant delay in diagnosis and treatment owing to these types of atypical presentation, particularly as hyperthermia and muscle rigidity may develop slowly or over several days. So great emphasis should be put on supportive tests such as creatinine phosphokinase levels and urine myoglobin.
Various laboratory parameters of the patient at the time of admission and discharge.
Footnotes
Consent statement
A well informed and written consent was taken from the patient.
Declaration of conflicting interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
