Hematopoietic stem cell transplantation for pulmonary alveolar proteinosis associated with primary immunodeficiency disease

Int J Hematol. 2018 May;107(5):610-614. doi: 10.1007/s12185-017-2375-1. Epub 2017 Nov 28.

Abstract

Pulmonary alveolar proteinosis (PAP) is a rare disorder that is characterized by the excessive accumulation of surfactant-like materials in the alveoli, leading to hypoxemic respiratory failure. We describe two Japanese infants with PAP associated with hypogammaglobulinemia and monocytopenia. These patients may have underlying primary immunodeficiency (PID) and were successfully treated with allogeneic hematopoietic stem cell transplantation (HSCT). This report indicates that allogeneic HSCT may provide a curative treatment for PAP associated with PID.

Keywords: Bronchoalveolar lavage; Granulocyte–macrophage colony stimulating factor; Hematopoietic stem cell transplantation; Primary immunodeficiency; Pulmonary alveolar proteinosis.

Publication types

  • Case Reports

MeSH terms

  • Agammaglobulinemia / etiology
  • Allografts
  • Asian People
  • Female
  • Granulocyte-Macrophage Colony-Stimulating Factor
  • Hematopoietic Stem Cell Transplantation*
  • Humans
  • Immunologic Deficiency Syndromes / complications*
  • Infant
  • Infant, Newborn
  • Leukopenia / etiology
  • Monocytes
  • Pulmonary Alveolar Proteinosis / etiology*
  • Pulmonary Alveolar Proteinosis / therapy*
  • Treatment Outcome

Substances

  • Granulocyte-Macrophage Colony-Stimulating Factor