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PPCM and type II autoimmune polyglandular syndrome

Published:April 24, 2018DOI:https://doi.org/10.1016/j.ajem.2018.04.053
      While it is true that “there is no single diagnostic test for peripartum cardiomyopathy (PPCM)” [
      • Sonmez B.M.
      • Iscanlt M.D.
      • Durdu T.
      • Kala I.
      • Tarhan N.
      • Uysal P.
      What is wrong with this postpartum woman?.
      ], over and above echocardiographic evaluation [
      • Sonmez B.M.
      • Iscanlt M.D.
      • Durdu T.
      • Kala I.
      • Tarhan N.
      • Uysal P.
      What is wrong with this postpartum woman?.
      ], patients with this diagnosis should also receive the benefit of endocrinological evaluation. The rationale is that PPCM may occur in association with either anterior pituitary failure [
      • Jalal I.
      • Desai R.K.
      • Maharaj I.C.
      • Pala A.S.
      • Joubert S.M.
      Isolated adrenocorticotrophic hormone (ACTH) deficiency associated with acute adrenal crisis.
      ,
      • Parikh A.
      • Ezzat S.
      Complete anterior pituitary failure and postpartum cardiomyopathy.
      ,
      • Wang S.-Y.
      • Hsu S.-R.
      • Su S.-L.
      • Tu S.-T.
      Sheehan's syndrome presenting with early postpartum congestive heart failure.
      ] or primary hypoadrenalism [
      • Jensen M.D.
      • Handwerger B.S.
      • Scheithauer B.W.
      • Carpenter P.C.
      • Mirakian R.
      • Banks P.M.
      Lymphocytic hypophysitis with isolated corticotropin deficiency.
      ], arguably, in some cases, on the basis of autoimmunity. One of the earliest examples was a 37 year old woman in whom isolated adrenocorticotrophic hormone (ACTH) deficiency coexisted with PPCM, the latter occurring two months after uneventful child birth [
      • Jalal I.
      • Desai R.K.
      • Maharaj I.C.
      • Pala A.S.
      • Joubert S.M.
      Isolated adrenocorticotrophic hormone (ACTH) deficiency associated with acute adrenal crisis.
      ]. A year after the publication of that case report isolated ACTH deficiency was reported as a manifestation of postpartum lymphocytic hypophysitis [
      • Jensen M.D.
      • Handwerger B.S.
      • Scheithauer B.W.
      • Carpenter P.C.
      • Mirakian R.
      • Banks P.M.
      Lymphocytic hypophysitis with isolated corticotropin deficiency.
      ]. Lymphocytic hypophysitis was also presumed to be the associated disorder in a 37 year old woman with PPCM and complete anterior pituitary failure who presented a week after normal and uneventful child birth [
      • Parikh A.
      • Ezzat S.
      Complete anterior pituitary failure and postpartum cardiomyopathy.
      ]. An autoimmune basis for PPCM is suggested by the fact that sera from PPCM patients contain high titers of autoantibodies against normal cardiac tissue proteins 37, 33, and 25 kD that are not present in the sera of patients with idiopathic cardiomyopathy [
      • Ansari A.A.
      • Fett J.D.
      • Carraway R.E.
      • Mayne A.E.
      • Onlamoon N.
      • Sundstrom J.B.
      Autoimmune mechanisms as the basis for human peripartum cardiomyopathy.
      ]. Therefore, according to one view, it might be hypothesised that a patient with PPCM has “a severe autoimmune condition … affecting multiple organs concomitantly” [
      • Parikh A.
      • Ezzat S.
      Complete anterior pituitary failure and postpartum cardiomyopathy.
      ]. However, even when pituitary failure is of presumed ischaemic aetiology, as is the case after postpartum haemorrhage, PPCM may be the outcome [
      • Wang S.-Y.
      • Hsu S.-R.
      • Su S.-L.
      • Tu S.-T.
      Sheehan's syndrome presenting with early postpartum congestive heart failure.
      ].
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