Indian J Pediatr DOI 10.1007/s12098-013-1323-1
SCIENTIFIC LETTER
A Case of Neuroleptic Malignant Syndrome in an Infant Lalitha Kailas & Sheeja Sugunan & G. S. Bindu & Christy Cathreen Thomas
Received: 24 July 2013 / Accepted: 23 December 2013 # Dr. K C Chaudhuri Foundation 2014
To the Editor: Neuroleptic malignant syndrome (NMS) is a rare adverse reaction to neuroleptics and other dopamine modulating agents [1]. It is an acute disorder of thermoregulation and neuromotor control due to idiosyncratic response to dopamine (D2) receptor blockade. Antipsychotic drugs that act by blockade of dopaminergic receptors are commonly implicated in most reported cases of NMS. Cases due to other drugs like domperidone, ketamine etc. have been reported, but this is the first case as per literature to be reported in a young infant due to domperidone [2]. A 4-mo-old boy, with single dysplastic kidney presented with febrile urinary tract infection (UTI). His urine culture grew E. coli sensitive to pipperacillin – tazobactum and hence the child was started on it. Child also had associated cough and mild breathlessness. His investigations at admission showed a deranged renal function test with blood urea of 40 mg/dL and serum creatinine of 1.5 mg/dL. His serum calcium was 9.6 mg/dL. Child showed some improvement to treatment initially with disappearance of fever by 48 h after starting antibiotic. As the child was getting intermittent worsening of breathlessness often following feed, gastroesophageal reflux disease was suspected and was started on domperidone 0.1 mg/kg/dose thrice daily with lanzoprazole and other supportive measures. Two days after starting domperidone child developed high grade continuous fever which was not responding to antipyretics with rigidity and tremor of both upper and lower limbs, irritability, sweating, L. Kailas : S. Sugunan (*) : G. S. Bindu : C. C. Thomas Department of Pediatrics, SAT Hospital, Government Medical College, Thiruvananthapuram, India e-mail: [email protected]
tachypnea and tachycardia. Investigations at this time revealed a total leucocyte count of 16,500/cmm. His alanine transaminase (ALT) was 143 IU/L and AST 122 IU/L. He also had acute deterioration of renal functions with blood urea rising to 133 mg/ dL and serum creatinine to 3.7 mg/dL. His total calcium had decreased to 4.6 mg/dL and potassium to 2.7 meq/L. Child also had elevated creatine phosphokinase (CPK) (2766 IU/L) and his urine was positive for myoglobin. The blood culture and repeat urine culture were sterile. His chest X-ray was unremarkable and cerebrospinal fluid (CSF) study, echocardiography and EEG were also normal. Serum parathormone was high (190 pg/mL). With the above clinical and laboratory findings a provisional diagnosis of neuroleptic malignant syndrome was made as he had all the 3 major (fever, rigidity and elevated creatine kinase) and 5 minor criteria (diaphoresis, changes in the level of consciousness, tachypnea, tachycardia and leucocytosis) for diagnosis of NMS [3]. Domperidone was discontinued and child was started on injection lorazepam with other supportive measures like IV fluids for proper hydration, urine alkalinisation, calcium and potassium supplementation etc. High temperature which was unresponsive to antipyretics before showed a rapid fall within 48 h of starting treatment. The defervesence was without antipyretics or change in antibiotics. Rigidity and tachypnea also showed gradual improvement with reduction in myoglobinuria, hypocalcemia and hypokalemia. By day four after initiation of treatment his blood urea was 40 mg/dL, serum creatinine 1.3 mg/dL, serum calcium 7.5 mg/dL and his urine was negative for myoglobin. CPK had also decreased to 900 IU/L. He showed complete resolution of all symptoms of NMS by six days after initiation of treatment. He was not rechallenged with domperidone.
Indian J Pediatr
A high index of suspicion is needed to diagnose NMS in children who are not on neuroleptics. This is the first case to be reported in a young infant due to domperidone [4, 5]. NMS even though rare in children should be considered in children who present with the typical features as delay in diagnosis and improper treatment may be fatal. Contributions All authors contributed to management, review of literature and drafting of the manuscript. SS will act as the guarantor for the paper. Conflict of Interest None. Role of Funding Source None.
References 1. Caroff SN, Mann SC. Neuroleptic malignant syndrome. Med Clin North Am. 1993;77:185–202. 2. Spirt MJ, Chan W, Thieberg M, Sachar DB. Neuroleptic malignant syndrome induced by domperidone. Dig Dis Sci. 1992;37:946–8. 3. Levenson JL. Neuroleptic malignant syndrome. Am J Psychiatry. 1985;142:1137–45. 4. Neuhut R, Lindenmayer JP, Silva R. Neuroleptic malignant syndrome in children and adolescents on atypical antipsychotic medication: A review. J Child Adolesc Psychopharmacol. 2009;19:415–22. 5. Silva RR, Munoz DM, Alpert M, Perlmutter IR, Diaz J. Neuroleptic malignant syndrome in children and adolescents. J Am Acad Child Adolesc Psychiatry. 1999;38:187–94.
SCIENTIFIC LETTER
A Case of Neuroleptic Malignant Syndrome in an Infant Lalitha Kailas & Sheeja Sugunan & G. S. Bindu & Christy Cathreen Thomas
Received: 24 July 2013 / Accepted: 23 December 2013 # Dr. K C Chaudhuri Foundation 2014
To the Editor: Neuroleptic malignant syndrome (NMS) is a rare adverse reaction to neuroleptics and other dopamine modulating agents [1]. It is an acute disorder of thermoregulation and neuromotor control due to idiosyncratic response to dopamine (D2) receptor blockade. Antipsychotic drugs that act by blockade of dopaminergic receptors are commonly implicated in most reported cases of NMS. Cases due to other drugs like domperidone, ketamine etc. have been reported, but this is the first case as per literature to be reported in a young infant due to domperidone [2]. A 4-mo-old boy, with single dysplastic kidney presented with febrile urinary tract infection (UTI). His urine culture grew E. coli sensitive to pipperacillin – tazobactum and hence the child was started on it. Child also had associated cough and mild breathlessness. His investigations at admission showed a deranged renal function test with blood urea of 40 mg/dL and serum creatinine of 1.5 mg/dL. His serum calcium was 9.6 mg/dL. Child showed some improvement to treatment initially with disappearance of fever by 48 h after starting antibiotic. As the child was getting intermittent worsening of breathlessness often following feed, gastroesophageal reflux disease was suspected and was started on domperidone 0.1 mg/kg/dose thrice daily with lanzoprazole and other supportive measures. Two days after starting domperidone child developed high grade continuous fever which was not responding to antipyretics with rigidity and tremor of both upper and lower limbs, irritability, sweating, L. Kailas : S. Sugunan (*) : G. S. Bindu : C. C. Thomas Department of Pediatrics, SAT Hospital, Government Medical College, Thiruvananthapuram, India e-mail: [email protected]
tachypnea and tachycardia. Investigations at this time revealed a total leucocyte count of 16,500/cmm. His alanine transaminase (ALT) was 143 IU/L and AST 122 IU/L. He also had acute deterioration of renal functions with blood urea rising to 133 mg/ dL and serum creatinine to 3.7 mg/dL. His total calcium had decreased to 4.6 mg/dL and potassium to 2.7 meq/L. Child also had elevated creatine phosphokinase (CPK) (2766 IU/L) and his urine was positive for myoglobin. The blood culture and repeat urine culture were sterile. His chest X-ray was unremarkable and cerebrospinal fluid (CSF) study, echocardiography and EEG were also normal. Serum parathormone was high (190 pg/mL). With the above clinical and laboratory findings a provisional diagnosis of neuroleptic malignant syndrome was made as he had all the 3 major (fever, rigidity and elevated creatine kinase) and 5 minor criteria (diaphoresis, changes in the level of consciousness, tachypnea, tachycardia and leucocytosis) for diagnosis of NMS [3]. Domperidone was discontinued and child was started on injection lorazepam with other supportive measures like IV fluids for proper hydration, urine alkalinisation, calcium and potassium supplementation etc. High temperature which was unresponsive to antipyretics before showed a rapid fall within 48 h of starting treatment. The defervesence was without antipyretics or change in antibiotics. Rigidity and tachypnea also showed gradual improvement with reduction in myoglobinuria, hypocalcemia and hypokalemia. By day four after initiation of treatment his blood urea was 40 mg/dL, serum creatinine 1.3 mg/dL, serum calcium 7.5 mg/dL and his urine was negative for myoglobin. CPK had also decreased to 900 IU/L. He showed complete resolution of all symptoms of NMS by six days after initiation of treatment. He was not rechallenged with domperidone.
Indian J Pediatr
A high index of suspicion is needed to diagnose NMS in children who are not on neuroleptics. This is the first case to be reported in a young infant due to domperidone [4, 5]. NMS even though rare in children should be considered in children who present with the typical features as delay in diagnosis and improper treatment may be fatal. Contributions All authors contributed to management, review of literature and drafting of the manuscript. SS will act as the guarantor for the paper. Conflict of Interest None. Role of Funding Source None.
References 1. Caroff SN, Mann SC. Neuroleptic malignant syndrome. Med Clin North Am. 1993;77:185–202. 2. Spirt MJ, Chan W, Thieberg M, Sachar DB. Neuroleptic malignant syndrome induced by domperidone. Dig Dis Sci. 1992;37:946–8. 3. Levenson JL. Neuroleptic malignant syndrome. Am J Psychiatry. 1985;142:1137–45. 4. Neuhut R, Lindenmayer JP, Silva R. Neuroleptic malignant syndrome in children and adolescents on atypical antipsychotic medication: A review. J Child Adolesc Psychopharmacol. 2009;19:415–22. 5. Silva RR, Munoz DM, Alpert M, Perlmutter IR, Diaz J. Neuroleptic malignant syndrome in children and adolescents. J Am Acad Child Adolesc Psychiatry. 1999;38:187–94.
