Hemophagocytic lymphohistiocytosis with granulomatosis and diffuse T-cell infiltration associated with disseminated Nocardiosis and pulmonary infection due to Streptomyces spp

• Published in: La revue de medecine interne Vol. 40; no. 7; pp. 457 - 461
• Main Authors: Canouï, E, Ingen-Housz-Oro, S, Ortonne, N, Lebeaux, D, Rodriguez-Nava, V, Godeau, B, Mahévas, M
• Format: Journal Article
• Language: French
• Published: France 01.07.2019
• Subjects: Aged; Chemotaxis, Leukocyte - physiology; Coinfection - diagnosis; Coinfection - immunology; Diagnosis, Differential; Female; Gram-Positive Bacterial Infections - complications; Gram-Positive Bacterial Infections - diagnosis; Gram-Positive Bacterial Infections - microbiology; Granuloma, Respiratory Tract - diagnosis; Granuloma, Respiratory Tract - microbiology; Humans; Lymphohistiocytosis, Hemophagocytic - complications; Lymphohistiocytosis, Hemophagocytic - diagnosis; Lymphohistiocytosis, Hemophagocytic - microbiology; Macrophage Activation Syndrome - diagnosis; Macrophage Activation Syndrome - microbiology; Nocardia - isolation & purification; Nocardia - pathogenicity; Nocardia Infections - complications; Nocardia Infections - diagnosis; Respiratory Tract Infections - diagnosis; Respiratory Tract Infections - microbiology; Skin Diseases, Bacterial - diagnosis; Skin Diseases, Bacterial - microbiology; Streptomyces - isolation & purification; Streptomyces - pathogenicity; T-Lymphocytes - immunology; T-Lymphocytes - physiology; granulomatosis; Nocardia; Syndrome d’activation lympho-histiocytaire; Hemophagocytic Lymphohistiocytosis; Granulomatose
• ISSN: 1768-3122
• DOI: 10.1016/j.revmed.2019.04.013

Hemophagocytic lymphohistiocytosis (HLH) is a rare syndrome frequently secondary to infectious disease, especially in immuno-compromised patients. We report a HLH secondary to disseminated nocardiosis and Streptomyces spp pulmonary infection. A 69-years-old women had recent subcutaneous nodules of the forearms and loins associated with peripheral neuropathy and pulmonary nodule of the right upper lobe. Cutaneous biopsy revealed granuloma. Cutaneous lesions worsened and the patient developed a HLH with probable cardiac and neurological involvement, associated with cutaneous granulomatosis and diffuse polyclonal lymphocyte proliferation. Nocardia PCR was positive in cutaneous biopsy. Pulmonary samples revealed Streptomyces in culture and Nocardia in PCR. The evolution under antibiotic treatment was favorable. Recent diagnosis of HLH without obvious etiology should lead to etiological investigation, including the search for infections with slow-growing bacteria such as Nocardia or Streptomyces spp.