Isoimmune neonatal neutropenia caused by Fc gamma RIIIb antibodies in a Spanish child

Puig, N; Haas, M. de; Kleijer, Marion; Montoro, J. A.; Pérez, Ana B.; Villalba, J.; Gómez, I.; Borne, A. E. G. K. Von Dem

doi:10.1046/j.1537-2995.1995.35895357901.x

Cited by 15 publications

(10 citation statements)

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“…A triallelic polymorphism on FcRIIIA has recently been identified (De Haas et al, 1996), whereas for FcRIIIB a biallelic, codominant neutrophil antigen (NA) system has been characterized (Werner et al, 1986;Huizinga et al, 1990b). The NA system is involved in clinical conditions such as neonatal alloimmune neutropenia and blood transfusion reactions (Huizinga et al, 1990c;Puig et al, 1995). Gene frequencies in a Caucasian population are 0 .…”

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confidence: 99%

NA‐phenotype‐dependent differences in neutrophil FcγRIIIb expression cause differences in plasma levels of soluble FcγRIII

Koene¹,

Haas²,

Kleijer³

et al. 1996

Br J Haematol

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Soluble Fc gamma RIII in plasma is primarily derived from neutrophils and is a measure of the total body neutrophil mass. We have developed a new, sensitive 'sandwich' ELISA to measure soluble Fc gamma RIII in plasma and released Fc gamma RIII in cell supernatants. Both sFc gamma RIIIa, derived from NK cells and sFc gamma RIIIb, derived from neutrophils are detected in the assay. However, plasma analysis of Fc gamma RIIIB gene-deficient donors suggested that sFc gamma RIIIa contributes only marginally to the total amount measured in healthy individuals. Furthermore, we observed that plasma of homozygous NA1-positive donors contained lower amounts of sFc gamma RIII than plasma of homozygous NA2-positive donors. Heterozygous donors were found to have intermediate levels of sFc gamma RIII in their plasma. Hemizygous Fc gamma RIIIB gene-deficient donors were found to have half the amount of sFc gamma RIII in their plasma compared to donors with two Fc gamma RIIIB alleles. These NA phenotype-dependent differences in plasma sFc gamma RIII could not be contributed to either an assay artefact or NA-dependent differences in shedding of Fc gamma RIIIb upon neutrophil activation. Calibration curves constructed with plasma of homozygous donors did nor reveal NA-dependent differences in antibody affinity. Measurement of released Fc gamma RIIIb in supernatants of neutrophils stimulated with PMA, and inhibition of this signal with human IgG revealed no NA-dependent differences. However, NA-dependent differences in neutrophil Fc gamma RIIIb expression were present, comparable to the differences found in plasma levels of sFc gamma RIII. Differences in the amounts of released Fc gamma RIII in supernatants of NA-typed apoptotic neutrophils were similar to initial differences in Fc gamma RIIIb expression, again being lower in NA1-positive than in heterozygous and NA2-positive donors. In conclusion, NA-dependent differences in plasma levels of soluble Fc gamma RIII seem to be caused by differences in expression of the receptor on the neutrophil membrane.

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confidence: 99%

NA‐phenotype‐dependent differences in neutrophil FcγRIIIb expression cause differences in plasma levels of soluble FcγRIII

Koene¹,

Haas²,

Kleijer³

et al. 1996

Br J Haematol

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“…Anti-CD16b has not been reported to confer a more significant or prolonged form of neonatal neutropaenia. 27,28 We acknowledge the limitations with our methodology. The rarity of NAIN necessitated the case series' design and inconsistencies in investigation between infants could not be controlled, including initiation, frequency and duration of ANC monitoring and the decision to undertake NAIN work-up.…”

Section: Discussionmentioning

confidence: 99%

Neonatal alloimmune neutropaenia: Experience from an Australian paediatric health service

Doan

Radhakrishnan

Krishnamurthy

et al. 2019

J Paediatrics Child Health

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Aim: To describe the presenting features and investigation findings in infants diagnosed with neonatal alloimmune neutropaenia (NAIN) within an Australian paediatric health network. The secondary aim was to describe the management and resolution of neutropaenia in infants with NAIN. Methods: A retrospective cohort study was conducted at Monash Children's Hospital, Melbourne, Australia. Infants referred to the Victorian Transplantation and Immunogenetics Service for evaluation of NAIN were identified and medical records were reviewed. Descriptive statistical analysis of infants' clinical outcomes, investigation findings and management was performed. Results: Nine infants were diagnosed with NAIN between December 2004 and June 2017. Overall incidence of NAIN was around 1 per 10 000 births. Median gestational age was 38 (range 35-40) weeks and birthweight was 2920 (2300-4445) g. Median age at NAIN work-up was 7 (2-33) days. Prior to investigation for NAIN, median absolute neutrophil count was 0.2 (0.01-0.6) × 10 9 cells/L. The post-natal ward was the source of presentation in most infants (78%). All except one infant were admitted to a neonatal unit and commenced on intravenous antibiotics (89%). Six infants were asymptomatic but received antibiotics for risk of infection (75%). Granulocyte-colony stimulating factor was administered to 44% of infants due to neutropaenia with presumed or confirmed infection. NAIN resolved at median age of 32 (6-200) days. Conclusions: Infants with NAIN frequently presented with severe, unexpected neutropaenia without major infection. Intravenous antibiotic therapy and granulocyte-colony stimulating factor use was common in this cohort. †Three pairs of siblings were observed: cases 2 and 4, and cases 6 and 7, respectively. Cases 8 and 9 were MCDA twins. EOI, early-onset infection, suspected or confirmed within 72 h of age; G-CSF, granulocyte-colony stimulating factor; ITR, immature-to-total neutrophil ratio (elevated if >0.2); LOI, late-onset infection, suspected or confirmed at or beyond 72 h of age; PROM, prolonged rupture of membranes (duration of rupture greater than 18 h); SCN, special care nursery.Neonatal alloimmune neutropaenia J Doan et al.

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“…Several cases of immune neonatal neutropenia caused by anti-Fc gamma RIIIb isoantibodies have been described in literature [4, 16–19]. Fromont et al report on the presence of iso-anti-CD-16 antibodies in one of five individuals with Fc gamma RIIIb deficiency (three of them pregnant women), all of them being healthy [17].…”

Section: Discussionmentioning

confidence: 99%

A Case of Neonatal Neutropenia Due to Anti-Fc Gamma Receptor IIIb Isoantibodies Treated with Recombinant Human Granulocyte Colony Stimulating Factor

Tomičić

Starčević²,

Zach³

et al. 2009

Case Reports in Medicine

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Alloimmunization to granulocyte-specific antigens can occur during pregnancy. Maternal antibodies of IgG class can cross the placenta to result in alloimmune neonatal neutropenia. Antibodies to human neutrophil antigens anti-HNA-1a, HNA-1b, and HNA-2a have been most commonly reported to cause alloimmune neonatal neutropenia. Isoantibodies to Fc gamma RIIIb (CD16) if mother is a HNA-null phenotype are rarely involved in neonatal neutropenia. We report on a case of severe neutropenia (440 neutrophils/μL) due to anti-Fc gamma RIIIb (CD16) isoimmunization. On day 14 severe omphalitis developed, which was treated for 7 days by an antibiotic (ceftriaxone in a dose of 80 mg/kg/d) according to umbilical swab finding. Omphalitis persisted for 10 days in spite of antibiotic therapy and only resolved upon the introduction of rhG-CSF therapy. Therapy with rh-GCSF proved efficient and led to neutrophil count increase to 1970/μL and cure of omphalitis. However, therapeutic effect on granulocyte count was of transient nature, as granulocyte count fell to 760 n/μL on day 4 of therapy discontinuation. Neutropenia persisted for 2 months. The newborn was discharged from the hospital on day 26 with normal clinical status with clinical and laboratory control examinations at 2-week intervals. No additional infections were observed during the course of neutropenia.

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Isoimmune neonatal neutropenia caused by Fc gamma RIIIb antibodies in a Spanish child

Cited by 15 publications

References 15 publications

NA‐phenotype‐dependent differences in neutrophil FcγRIIIb expression cause differences in plasma levels of soluble FcγRIII

NA‐phenotype‐dependent differences in neutrophil FcγRIIIb expression cause differences in plasma levels of soluble FcγRIII

Neonatal alloimmune neutropaenia: Experience from an Australian paediatric health service

A Case of Neonatal Neutropenia Due to Anti-Fc Gamma Receptor IIIb Isoantibodies Treated with Recombinant Human Granulocyte Colony Stimulating Factor

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