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1 Unité d'Immunologie et Hématologie Pédiatrique, Institut National de la Santé et de la Recherche Médicale(INSERM) U429, Paris, France
2 Unité d'Immunologie et Hématologie Pédiatrique, Paris, France
3 Service d'Anatomopathologie, CHU Necker Enfants Malades, Paris, France
4 Institut National de la Santé et de la Recherche Médicale(INSERM) U429, Paris, France
5 Service de Pédiatrie, CHU, St Etienne, Paris France
6 Service de Pédiatrie, CHIC, Créteil, Paris ,France
7 Service de Pédiatrie, CHU, Nancy, Paris, France
8 Service de Pédiatrie, CHU, Lille, Paris, France
9 Service de Pédiatrie, CHU Edouard Herriot, Lyon, Paris, France
10 Service de Pédiatrie, CHU, Nice, Paris, France
11 Service de Pédiatrie, CHU Robert Debré, Paris, France
12 Service de Pédiatrie, CHU Kremlin-Bicêtre, Bicêtre, Paris, France
13 Service d'Hématologie, CHU Bichat, Paris, France
14 Service de Microbiologie, CHU Necker Enfants Malades, Paris, France
Objective. Disseminated bacillus Calmette Guérin (BCG) infection after inoculation of live vaccine is considered to result from impaired immunity of the child. However, in half of the cases, regarded as idiopathic, no well-defined immunodeficiency condition can account for the infection. The objective of the present study is to report the prevalence, clinical features, associated infections, and outcomes of children with idiopathic disseminated BCG infection.
Design. National retrospective survey during the period from 1974 through 1994 in France.
Setting. All neonatology and pediatrics units in primary care and referral centers throughout France.
Patients. Data were collected from 595 (82%) of 721 units, 377 (93%) of 407 centers, and 320 (93%) of 345 cities. Selection criteria included BCG infection, dissemination to at least two areas beyond the inoculation site, and no well-defined immunodeficiency condition. Sixteen children (8 girls and 8 boys), born to families unrelated to each other but often consanguinous (5 of 16), satisfied the criteria; children were born in France (11 of 16) or abroad (5 of 16).
Results. The minimal prevalence rate was estimated at 0.59 cases per 1 million vaccinated children born in France. Clinical features included fever and cachexia, disseminated BCG infection to lymph nodes (15 of 16), skin (13 of 16), soft tissues (11 of 16), lungs (11 of 16), spleen (11 of 16), liver (11 of 16), and bones (9 of 16). Eight children had associated or subsequent severe opportunistic infection (50%), with either nontyphi Salmonella enterica serotypes (7 of 16) or Mycobacterium abscessus (1 of 16). The outcome was poor: 8 children (50%) died; the cause of death was BCG infection for most children (7 of 8); 8 survived until the last follow-up (50%).
Conclusions. Idiopathic disseminated BCG infection is a rare but severe complication of BCG vaccination. The infection probably results from an as yet unknown genetically determined immunodeficiency condition that affects the killing of intracellular bacteria such as BCG and Salmonella.
Submitted on April 4, 1996
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