Abstract Non 01, Non O139 Vibrio cholerae bacteraemia is a rare but potentially fatal occurrence. There have been very few incidents of this infection from around the world. The treatment regimen of antibiotics also varies in literature. We present a case of bacteraemia caused by Non O1, Non O139 Vibrio cholerae along with associated risk factors, disease manifestations, laboratory diagnosis and treatment regimen. This serves to add additional information regarding symptoms and signs of this infection along with management of patient. Knowledge regarding this topic shall be highly useful to professionals if further cases are detected. In the discussion section, a review of literature of previous cases is also presented. Keywords: Vibrio cholera, Bacteraemia, Non O1, Non O139, Pakistan. Introduction Vibrio cholerae is a gram negative oxidase positive bacterium. Its O1 and O139 serotypes are notorious for causing epidemics and pandemics of severe watery diarrhoea known as cholera. Strains that do not agglutinate with O1 and O139 antisera are known as non O1, non O139 Vibrio cholerae species. These strains — although can also cause diarrhoeal illness, are reputable more due to their extra intestinal manifestations — most prominent of which is bacteraemia. Non O1, non O139 bacteraemia is rare and very few cases of it in infants has been presented in the previous literature. Case Report A two-month old baby boy presented with a 10-day history of fever and abdominal distension. He had abdominal pain for the past 3 days and was taken to a local doctor who administered a single dose of metronidazole. However, the condition of the baby worsened and the infant became lethargic. After which he was subsequently brought to a tertiary care hospital. Within a span of one month he was admitted thrice. He had an uneventful antenatal history with an emergency caesarian section. He was born with bilateral lamellar cataract. His birth weight was 3.5 kg and he had neonatal jaundice on the third day of birth. On 19th day his weight increased by 200grams. On his third visit, on examination he was found to be drowsy with a temperature of 38°C, a heart rate of 160 beats per minute and a respiratory rate of 35 breaths per minute. His oxygen saturation was 98 percent. He also appeared anaemic and had jaundice along with pitting oedema. On Cardiovascular system examination S1 and S2 were audible while pulses and perfusion were poor. Glasgow coma scale revealed a 14/15 score. Abdomen was tense, distended with a positive fluid thrill. He also had hepatomegaly, splenomegaly, oedematous skin with an everted umbilicus and sluggish bowel sounds. Chest X ray was clear. The patient was started on cefotaxime, amikacin and vitamin K and a nasogastric tube was inserted. Ultrasound showed dilated bowel loops. Blood cultures were sent. Gram stain revealed curved gram negative rods. After 24 hours of incubation, growth of beta haemolytic colonies were noted on sheep blood agar. On MacConkey agar there was growth of non-lactose fermenters that were oxidase positive. Hanging drop showed darting movements. On Thiosulfate- citrate- bile salts- sucrose (TCBS) agar there was growth of yellow colonies that were opaque in center and pale at periphery. API-20E (bioMérieux) confirmed identification of Vibrio cholera while slide agglutination tests were negative for O1 and O139 antigens. On third day of admission the patient started to have symptoms of respiratory distress. Abdominal swelling increased and patient became drowsy with laboured breathing. Oxygen saturation reduced to 84% from 95%. Parents were advised for intubation. However, they refused and due to poor prognosis, withdrew support. Consent from parents was taken prior to the writing of the J Pak Med Assoc 650 CASE REPORT Non O1, non O139 Vibrio cholerae bacteraemia in an infant; case report and literature review Mirza Zain Baig, 1 Umme Hani Abdullah, 2 Yusra Shafquat, 3 Khadija Nuzhat Humayun, 4 Afia Zafar 5 1,2 Medical College, 3,5 Section of Microbiology, Department of Pathology and Laboratory Medicine, 4 Department of Paediatrics and Child Health, Aga Khan University, Karachi. Correspondence: Afia Zafar. Email: afia.zafar@aku.edu