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2014 ; 93
(2
): 61-72
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Idiopathic CD4 lymphocytopenia: clinical and immunologic characteristics and
follow-up of 40 patients
#MMPMID24646462
Régent A
; Autran B
; Carcelain G
; Cheynier R
; Terrier B
; Charmeteau-De Muylder B
; Krivitzky A
; Oksenhendler E
; Costedoat-Chalumeau N
; Hubert P
; Lortholary O
; Dupin N
; Debré P
; Guillevin L
; Mouthon L
Medicine (Baltimore)
2014[Mar]; 93
(2
): 61-72
PMID24646462
show ga
Idiopathic CD4 T lymphocytopenia (ICL) is a rare and severe condition with
limited available data. We conducted a French multicenter study to analyze the
clinical and immunologic characteristics of a cohort of patients with ICL
according to the Centers for Disease Control criteria.We recruited 40 patients
(24 female) of mean age 44.2 ± 12.2 (19-70) years. Patients underwent
T-lymphocyte phenotyping and lymphoproliferation assay at diagnosis, and
experiments related to thymic function and interferon (IFN)-? release by natural
killer (NK) cell were performed. Mean follow-up was 6.9 ± 6.7 (0.14-24.3) years.
Infectious, autoimmune, and neoplastic events were recorded, as were outcomes of
interleukin 2 therapy.In all, 25 patients had opportunistic infections (12 with
human papillomavirus infection), 14 had autoimmune symptoms, 5 had malignancies,
and 8 had mild or no symptoms. At the time of diagnosis, the mean cell counts
were as follows: mean CD4 cell count: 127/mm (range, 4-294); mean CD8: 236/mm
(range, 1-1293); mean CD19: 113/mm (range, 3-547); and mean NK cell count: 122/mm
(range, 5-416). Most patients had deficiency in CD8, CD19, and/or NK cells.
Cytotoxic function of NK cells was normal, and patients with infections had a
significantly lower NK cell count than those without (p = 0.01). Patients with
autoimmune manifestations had increased CD8 T-cell count. Proliferation of thymic
precursors, as assessed by T-cell rearrangement excision circles, was increased.
Six patients died (15%). CD4 T-cell count <150/mm and NK cell count <100/mm were
predictors of death.In conclusion, ICL is a heterogeneous disorder often
associated with deficiencies in CD8, CD19, and/or NK cells. Long-term prognosis
may be related to initial CD4 and NK cell deficiency.
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