INTRODUCTION: A 5-month-old male infant presented with fever, vomiting, deterioration of consciousness and respiratory failure. He had a difficult intubation with multiple attempts and subsequently multiple failed extubations. Bronchoscopy revealed bilateral vocal cord paralysis, finally diagnosed as infant botulism.
CASE PRESENTATION: The patient was brought to a local community hospital with a history of increasing drowsiness. He had had 4 days of fever and 1 day of vomiting prior to admission. Physical examination revealed an afebrile infant with cold extremities and lethargy. His Glasgow Coma Scale was 6. He had tachycardia, tachypnea and respiratory distress. Lungs were clear to auscultation. He went on to develop respiratory failure and experienced cardiac arrest (of 23 minutes duration) due to hypoxia during a difficult intubation. Cardiovascular stability was maintained with a dopamine infusion prior to referral. Upon arrival, his Glasgow Coma Scale was 3 and significant hypotonia was present. His pupils were 5 mm, non-reactive. Broad spectrum antibiotics and acyclovir were administered. Initial investigations including complete blood count, urinalysis, electrolytes, renal function and liver function tests were unremarkable. Chest radiography, neuroimaging, and echocardiogram were all within normal limits. His blood, nasopharyngeal swab, non-bronchoscopic bronchoalveolar lavage and urine cultures were all negative. He was fully awake and alert on the third day after intubation. As he had a good cough and a gag reflex was excellent, the decision was made to extubate him on the 4th day of admission. Biphasic stridor, increased work of breathing and paradoxical movement of chest and abdomen were observed. He was re-intubated within 2 hours. Subsequent extubations were attempted on the 5th and the 7th days after admission. Neither attempt was successful. His neurological examination was within normal limits except he had a persistent mild truncal hypotonia. The repeat head and neck CT scan were unremarkable. Chest CT scan revealed only bibasilar and right apical subsegmental atelectasis. The ultrasonography of diaphragm revealed normal diaphragmatic movement. Flexible bronchoscopy was performed and bilateral vocal cord paralysis was discovered. Two weeks after admission, extubation was again attempted. The patient suddenly developed biphasic stridor and acute respiratory distress. The flexible bronchoscopy was performed and revealed immobilized left vocal cord and partial movement of the right vocal cord. The patient was re-intubated with a plan to re-evaluate in 1 week before considering tracheostomy. A successful extubation was performed on day 20 of admission. Stool Clostridium Botulinum toxin B came back positive. There was no history of honey or canned-food ingestion. The hypotonia was gradually improved without major neurological impairment. He was discharged home after 1 month of hospitalization.
DISCUSSIONS: Infant botulism is the most frequent form of human botulism caused by Clostridium botulinum which is colonized in small intestines before hematogenous spread. Most of affected infants presents between 1–6 months of age. The definite diagnosis can be made from positive botulism toxin or spores in stool. Enteric toxin causes intestinal immotility while neurotoxin blocks pre-synaptic Acetylcholine release, causing bulbar palsy, skeletal muscle weakness, flaccid paralysis and respiratory arrest. Most of the patients recovered without sequalae. Botulism immunoglobulin is now available and may be useful if it is given within 2 weeks after onset.
CONCLUSION: Botulism should be considered in an infant who presents with poor feeding, lethargy, weakness and respiratory failure.
DISCLOSURE: Harutai Kamalaporn, No Financial Disclosure Information; No Product/Research Disclosure Information