title: [Shifting cellulitis in a patient with X-linked hypogammaglobulinemia].
abstract: BACKGROUND
  In cases of immunodeficiency, a systemic infection may be revealed by atypical symptoms, particularly those involving the skin.
  
  
  METHODS
  The present case describes a 19-year-old male with X-linked hypogammaglobulinemia, or Bruton agammaglobulinemia, treated with intravenous immunoglobulin G antibodies. Over a 6-week period, the patient developed recurrent plaques in both legs, first on one and then on the other, without fever. Blood cultures were repeated and the fifth pair proved positive for Campylobacter jejuni. An abdominal scan showed appendicitis without intestinal signs. The patient was treated with azithromycin for 2 weeks, which resulted in full recovery from the skin lesions.
  
  
  CONCLUSIONS
  Campylobacter bacteremia infections are severe and carry a 15% mortality rate at 30 days. The majority of affected patients present humoral immunodeficiency. The literature contains reports of 10 patients with C. jejuni-associated cellulitis, of whom 6 presented hypogammaglobulinemia. We postulate that the cutaneous manifestations were caused by septic metastases. The immunoglobulin replacement therapy mainly comprised IgG antibodies; IgA and IgM antibodies appear to play a key role in the response to C. jejuni infection, which could explain the susceptibility observed. The American guidelines recommend blood and skin cultures in patients with cellular immune defects. We suggest that this recommendation be extended to patients with humoral immunodeficiency.
authors.affiliation: Hôpital Pontchaillou, service de dermatologie, CHU de Rennes, 2, rue Henri-Le-Guilloux, 35000 Rennes, France.
authors.forename: F
authors.initials: F
authors.lastname: Poizeau
authors.affiliation: Hôpital Pontchaillou, service de dermatologie, CHU de Rennes, 2, rue Henri-Le-Guilloux, 35000 Rennes, France; Université de Rennes 1, 2, avenue du Professeur-Léon-Bernard, 35043 Rennes cedex, France; Hôpital Pontchaillou, Inserm CIC 1414, unité de pharmacoépidémiologie, CHU de Rennes, 2, rue Henri-Le-Guilloux, 35033 Rennes cedex 9, France. Electronic address: catherine.droitcourt@chu-rennes.fr.
authors.forename: C
authors.initials: C
authors.lastname: Droitcourt
authors.affiliation: Hôpital Pontchaillou, service de dermatologie, CHU de Rennes, 2, rue Henri-Le-Guilloux, 35000 Rennes, France.
authors.forename: C
authors.initials: C
authors.lastname: Saillard
authors.affiliation: Hôpital Pontchaillou, service de dermatologie, CHU de Rennes, 2, rue Henri-Le-Guilloux, 35000 Rennes, France; Hôpital Sud, service de médecine interne, CHU de Rennes, 16, boulevard de Bulgarie, 35200 Rennes, France.
authors.forename: M
authors.initials: M
authors.lastname: Poirot
authors.affiliation: Hôpital Sud, service de médecine interne, CHU de Rennes, 16, boulevard de Bulgarie, 35200 Rennes, France.
authors.forename: T
authors.initials: T
authors.lastname: Le Gallou
authors.affiliation: Hôpital Sud, service de médecine interne, CHU de Rennes, 16, boulevard de Bulgarie, 35200 Rennes, France.
authors.forename: A
authors.initials: A
authors.lastname: Perlat
authors.affiliation: Hôpital Pontchaillou, service de dermatologie, CHU de Rennes, 2, rue Henri-Le-Guilloux, 35000 Rennes, France; Université de Rennes 1, 2, avenue du Professeur-Léon-Bernard, 35043 Rennes cedex, France; Hôpital Pontchaillou, unité de pharmacoépidémiologie, CHU de Rennes, 2, rue Henri-Le-Guilloux, 35033 Rennes cedex 9, France.
authors.forename: A
authors.initials: A
authors.lastname: Dupuy
mesh_terms: D000361:Agammaglobulinemia / Q000150:complications*; D000900:Anti-Bacterial Agents / Q000627:therapeutic use; D001064:Appendicitis / Q000382:microbiology; D017963:Azithromycin / Q000627:therapeutic use; D016470:Bacteremia / Q000188:drug therapy / Q000382:microbiology; D002169:Campylobacter Infections / Q000175:diagnosis* / Q000188:drug therapy; D002481:Cellulitis / Q000188:drug therapy / Q000382:microbiology*; D040181:Genetic Diseases, X-Linked / Q000150:complications*; D006801:Humans; D008297:Male; D055815:Young Adult
chemical_list: D000900:Anti-Bacterial Agents; D017963:Azithromycin
keywords: Bacteremia; Bactériémie; Bruton; Campylobacter; Cellulitis; Dermo-hypodermite bactérienne; Erysipela; Hypogammaglobulinemia; Hypogammaglobulinémie; Érysipèle