Primary intestinal lymphangiectasia (PIL) is rare disorder characterized by congenital malformation or obstruction of intestinal lymphatic drainage; it is responsible for protein losing enteropathy leading to lymphopenia, hypoalbuminemia and hypogammaglobulinemia. A low-fat diet associated with medium-chain triglyceride supplementation is the cornerstone of PIL management. The administration of intravenous immunoglobulins does not always lead to satisfactory plasma levels and therefore the replacement therapy with immunoglobulins is controversial. We describe here the case of a patient with PIL and severe hypogammaglobulinemia treated with immunoglobulins. The striking aspect of this case is the clinical and serological benefit obtained with the subcutaneous compared to the intravenous immunoglobulins administration.

Secondary hypogammaglobulinemia in Waldmann's disease treated with subcutaneous immunoglobulins

PATUZZO, Giuseppe;TINAZZI, Elisa;PUCCETTI, Antonio;LUNARDI, Claudio
2016-01-01

Abstract

Primary intestinal lymphangiectasia (PIL) is rare disorder characterized by congenital malformation or obstruction of intestinal lymphatic drainage; it is responsible for protein losing enteropathy leading to lymphopenia, hypoalbuminemia and hypogammaglobulinemia. A low-fat diet associated with medium-chain triglyceride supplementation is the cornerstone of PIL management. The administration of intravenous immunoglobulins does not always lead to satisfactory plasma levels and therefore the replacement therapy with immunoglobulins is controversial. We describe here the case of a patient with PIL and severe hypogammaglobulinemia treated with immunoglobulins. The striking aspect of this case is the clinical and serological benefit obtained with the subcutaneous compared to the intravenous immunoglobulins administration.
2016
Waldmann‘s disease; primary intestinal lymphangectasia; protein-losing enteropathy; secondary immunodeficiencies; subcutaneous immunoglobulins; Adult; Agammaglobulinemia; Diet, Fat-Restricted; Humans; Immunoglobulin G; Immunologic Factors; Infusions, Subcutaneous; Lymphangiectasis, Intestinal; Lymphedema; Male; Severity of Illness Index; Treatment Outcome; Triglycerides
File in questo prodotto:
File Dimensione Formato  
Secondary hypogammaglobulinemia in Waldmann’s disease treated with subcutaneous immunoglobulins.pdf

accesso aperto

Tipologia: Versione dell'editore
Licenza: Dominio pubblico
Dimensione 102.69 kB
Formato Adobe PDF
102.69 kB Adobe PDF Visualizza/Apri

I documenti in IRIS sono protetti da copyright e tutti i diritti sono riservati, salvo diversa indicazione.

Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11562/946759
Citazioni
  • ???jsp.display-item.citation.pmc??? 2
  • Scopus 9
  • ???jsp.display-item.citation.isi??? ND
social impact