Objective To report one case of neuroleptic malignant syndrome (NMS) with raising blood sugar. Methods The patient was studied clinically with biochemistry, white blood cells, psychiatric symptoms, and creatine phosphokinase (CPK) observations. Results The male patient with a history of taking antipsychotics more 30 years and his age of onset was about 20 years. He had severe muscular rigidity, altered consciousness and autonomic disturbance associated with elevation of serum CPK levels (max 3755 U/L) and leucocytosis(max 13.3×10 9 /L), especially granular leukocytosis( max 90%) and lymphocytopenia (min 8%). In addition, high blood sugar emerged along with the variation of white blood cells (max 9.0 mmol/L). Conclusion The manifestations of the patient was in conformity with those of the NMS. The patient had catatonic signs such as mutism, drinking difficulty, etc. and excess of saliva. Developmental observation with CPK and white blood cells is able to reveal the severity level of NMS. Raising blood sugar should be also monitoring item. Objective To report one case of neuroleptic malignant syndrome (NMS) with raising blood sugar. Methods The patient was studied clinically with biochemistry, white blood cells, psychiatric symptoms, and creatine phosphokinase (CPK) observations. Results The male patient with a history of taking He had severe muscular rigidity, altered consciousness and autonomic disturbance associated with elevation of serum CPK levels (max 3755 U / L) and leucocytosis (max 13.3 × 10 9 / L). Antipsychotics more 30 years and his age of onset was about 20 years. , especially granular leukocytosis (max 90%) and lymphocytopenia (min 8%). In addition, high blood sugar presenting along with the variation of white blood cells (max 9.0 mmol / L). Conclusion The manifestations of the patient was in conformity with those of the NMS. The patient had catatonic signs such as mutism, drinking difficulty, etc. and excess of saliva. Developmental observation with CPK and white blood cells is able to reveal the severity level of NM S. Raising blood sugar should be also monitoring item.