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2023

ANUÁRIO DO HOSPITAL
DONA ESTEFÂNIA

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SACROILIITIS BY CORINEBACTERIUM DIPHTHERIAE

Jorge Rodrigues1, Margarida Pinto2, Maria João Brito1, Alexandra Vasconcelos1, Paula Lavado3, Catarina Gouveia1

1 - Pediatric Infectious Diseases Unit, Hospital Dona Estefânia, Centro Hospitalar Universitário de Lisboa Central, EPE, Lisbon, Portugal
2 - Laboratory of Microbiology, Department of Clinical Pathology, Hospital Dona Estefânia, Centro Hospitalar Universitário de Lisboa Central, EPE, Lisbon, Portugal
3 - Department of Infectious Diseases, National Institute of Health Dr. Ricardo Jorge, Lisbon, Portugal

- Reunião internacional (38th meeting of the European Society for Paediatric Infectious Diseases). Publicação em versão integral no The Pediatric Infectious Disease Journal.

Resumo:
Introduction: Although potentially fatal, infection by Corynebacterium diphtheriae became rare in the developed world since the introduction of effective immunization. The burden of disease lies mainly in developing countries. Nevertheless, systemic infections have recently been increasing, mostly from emerging nontoxigenic strains with potential to cause invasive disease. These infections are often associated with travel to prevalent endemic areas of C. diphtheriae.
Case presentation: An incompletely immunized 10-year-old girl with corrected tetralogy of Fallot presented to the emergency department with right upper thigh pain which limited her walking associated with 7-day fever, vomiting and diarrhea. She had returned from rural Cape Verde where she contacted with various livestock and drank unsafe water. Blood tests revealed microcytic, hypochromic anemia, leukocytosis (17350/uL), thrombocytopenia, elevated C-reactive protein (355.8 mg/L), procalcitonin (5.05 ng/ml) and erythrocyte sedimentation rate (76 mm/h). Hip and pelvic radiography and hip ultrasonography were normal. Sacroiliitis was confirmed by MRI. Blood and fecal cultures were obtained and she started ceftriaxone and clindamycin. A nontoxigenic strain of Corynebacterium diphtheriae was isolated from 2 blood cultures and treatment was changed to penicillin. Cutaneous, otolaryngologic and cardiac involvement were excluded. On day 4, she developed signs of arthritis on her right ankle and underwent arthrocentesis, whose culture was negative. She completed 4 weeks of amoxicillin with favorable outcome. Remaining cultures were sterile. On discharge, she updated her immunization schedule (sixth dose of diphtheria-tetanus toxoid). Re-evaluated after 2, 4 and 8 weeks, she stayed asymptomatic.
Learning points: Corynebacterium diphtheriae remains a major human pathogen worldwide. Infection by nontoxigenic, highly pathogenic clones in Europe has been rising, although osteoarticular infection has rarely been reported. These strains are known to cause atypical course of disease, especially in predisposed individuals such as congenital heart disease. Despite lacking diphtheria vaccine booster, toxoid immunization still would not have protected our patient against nontoxigenic strains.

Palavras Chave: sacroiliitis, osteoarticular infection, endocarditis, Corynebacterium diphtheriae