Rituximab-induced remission of autoimmune hypophysitis and primary immune thrombocytopenia in a patient with autoimmune polyendocrine syndrome type 4

Abstract

Rituximab, a B-cell depleting antibody, has been used for treatment of several autoimmune diseases. We report the effect of rituximab therapy on pituitary and platelet autoimmunity in a 36-yr old patient, positive for antiplatelet and antipituitary (APA) antibodies. The behavior of pituitary function and of APA by immunofluorescence, as well antibodies to platelets and platelet count, were investigated at start and subsequently every six months during Rituximab treatment. Rituximab treatment determined disappearance of antiplatelet antibodies with recovery of normal platelet count and disappearance of APA with recovery of pituitary-gonadal function. Rituximab determined a remission of both autoimmune processes, likely through a T cell inactivation and a depletion of autoreactive B-cells generation responsible for antiplatelet and antipituitary antibody production.

Keywords::

• antibodies to platelets
• antipituitary antibodies
• autoimmune hypophysitis
• autoimmune hypopituitarism
• immune thrombocytopenia

Financial & competing interests disclosure

This work was supported in part by grants from University Research Grants 2010, Second University of Naples. The authors have no other relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript apart from those disclosed.

No writing assistance was utilized in the production of this manuscript.

Key issues

• Lymphocytic hypophysitis is frequently associated with other autoimmune endocrine and nonendocrine diseases.

• The most common association is with Hashimoto’s thyroiditis or Graves’ disease but is rarely associated with thrombocytopenic purpura.

• Many cases of lymphocytic hypophysitis could be included in complete type 3 autoimmune polyendocrine syndrome (autoimmune thyroid diseases without Addison disease).

• Rituximab (RTX), a B-cell-depleting antibody, has been used for the treatment of several autoimmune diseases.

• We report the effect of RTX therapy on pituitary and platelet autoimmunity in a 36-year-old patient, positive for antiplatelet and antipituitary (APA) antibodies.

• RTX treatment determined the disappearance of antiplatelet antibodies with recovery of normal platelet count and disappearance of APA with recovery of pituitary-gonadal function.

• Our case report may open the way for other further studies on more large population, which may allow to include RTX-associated disease treatment at doses utilized for therapy of one autoimmune disease, not only among the therapeutic options for LYH but also among those to be utilized in patients with two or more autoimmune diseases.