Human adenoviral (HAdV) chronic arthritis expands the infectious spectrum of primary agammaglobulinemia

Abstract

Inborn errors of immunity (IEI) are a heterogeneous entity with an increasing number of late diagnoses. Besides infections, inflammatory manifestations are a growing part of the clinical landscape of IEI. These complications are of unknown causes and often lead to the prescription of immunosuppressive agents that worsen the underlying immune defect. We here report the case of an adult patient diagnosed with chronic Human Adenovirus C-1 arthritis in the setting of primary agammaglobulinemia. Metagenomic next-generation sequencing led to the correct diagnosis and high-dose intravenous immunoglobulins resulted in complete recovery. This observation gives new insights into adenoviral immunity and underlines the importance of metagenomics in the diagnosis of inflammatory manifestations in immunocompromised patients.

Keywords: Adenoviral arthritis; Agammaglobulinemia; Metagenomic next generation sequencing; Inborn errors of immunity.

Fig. 1

A. Pedigree of the patient (depicted in black, III.3 or P1) with Human Adenoviral polyarthritis B. Chest Computed Tomography of P1 showing diffuse bronchiectasis. C. Bruton’s Tyrosine Kinase (Btk) expression in CD14 + monocytes and in rare CD19 + B-cells of P1 compared to a healthy control and to a patient with X-linked agammaglobulinemia (XLA or Bruton’s disease). D. Kinetics of HAdV DNA in synovial fluid (red), stools (green) and plasma (blue). Pink area correspond to timepoints of IVIg infusion