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PyodermaGangrenosum–LikeUlcer in a PatientWith X-LinkedAgammaglobulinemia Patrick R. MurrayAshish JainGülbû Uzel2010 год

Pyoderma Gangrenosum–Like Ulcer in a Patient With X-Linked Agammaglobulinemia
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Аннотация:

Background

Pyoderma gangrenosum–like ulcers and cellulitis of the lower extremities associated with recurrent fevers in patients with X-linked (Bruton) agammaglobulinemia have been reported to be caused byHelicobacter bilis(formerly classified asFlexispira rappiniand thenHelicobacterstrain flexispira taxon 8). Consistent themes in these reports are the difficulty in recovering this organism in blood and wound cultures and in maintaining isolates in vitro. We confirmed the presence of this organism in a patient's culture by using a novel application of gene amplification polymerase chain reaction and electrospray ionization time-of-flight mass spectrometry.

Observation

An adolescent boy with X-linked agammaglobulinemia presented with indurated plaques and a chronic leg ulcer whose origin was strongly suspected to be anH bilisorganism. Histologic analysis demonstrated positive Warthin-Starry staining of curvilinear rods, which grew in culture but failed to grow when subcultured. They could not be identified by conventional techniques. A combination of gene amplification by polymerase chain reaction and electrospray ionization time-of-flight mass spectrometry confirmed the identity of this organism.

Conclusions

This novel technology was useful in the identification of a difficult-to-growHelicobacterorganism, the cause of pyoderma gangrenosum–like leg ulcers in patients with X-linked agammaglobulinemia. Correct identification of this organism as the cause of pyoderma gangrenosum–like ulcers in patients with X-linked agammaglobulinemia is of great importance for the early initiation of appropriate and curative antibiotic therapy.
Год издания: 2010
Авторы: Patrick R. Murray, Ashish Jain, Gülbû Uzel, Raymond Ranken, Cristina Ivy, Lawrence B. Blyn, David J. Ecker, Rangarajan Sampath, Chyi‐Chia Richard Lee, Maria L. Turner
Издательство: American Medical Association
Источник: Archives of Dermatology
Ключевые слова: Immunodeficiency and Autoimmune Disorders, Autoimmune and Inflammatory Disorders, Chronic Lymphocytic Leukemia Research
Другие ссылки: Archives of Dermatology (PDF)
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Europe PMC (PubMed Central) (PDF)
Europe PMC (PubMed Central) (HTML)
PubMed Central (HTML)
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