Postpartum thyroiditis and hypothalamo-hypophysial insufficiency in the same woman with successive pregnancies: A case report

Department of Endocrinology and Metabolic Diseases, Ufuk University Medical Faculty, Dr. Ridvan Ege Hospital, Ankara, Turkey.
Gynecological Endocrinology (Impact Factor: 1.33). 09/2009; 26(2):105-8. DOI: 10.3109/09513590903215532
Source: PubMed


Although the incidence of postpartum autoimmune disorders of endocrine glands are not rare, the presence of two different entities in the same patient with two different pregnancies is uncommon.
We present a 35-year-old woman whose story starts with her first pregnancy when she was 29 years old, she had the diagnosis of postpartum thyroiditis with hypothyroidism.We followed up the patient when she had her second pregnancy.
When she was being followed up with levothyroxine replacement, 5 years later she had her second delivery after which she had complaints of polydipsia, polyuria, weight loss and had the diagnosis of central diabetes insipitus and she has started desmopressin treatment and 17 months later the delivery she again applied with amenorrhea, continuation of lactation later she noticed oligomenorrhea, and her gonadotropin levels were found to be low as well as her TSH levels, although the L-thyroxine treatment dose was not changed. Dynamic tests of hypophysis revealed hypophyseal insufficiency and repeated hypophyseal MRI was in concordance with lymphocytic hypophysitis which explains the pattern of endocrinological abnormalities after the second delivery.
This case signals role of autoimmune mechanisms underlying the endocrinopathies seen after successive pregnancies of the same patient.

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    ABSTRACT: Background: Postpartum thyroiditis (PPT) is characterized by the development of postpartum thyroid dysfunction, which may occur up to 12 months after delivery. The syndrome usually presents with transient thyrotoxicosis, followed by transient hypothyroidism. The association of this condition with resistance to thyroid hormones (RTH) has never been described. Patient findings: In this report, we describe a 30-year-old patient affected by RTH due to a novel p.V283A thyroid hormone receptor-β (THRB) heterozygous mutation in exon 8, which affects the ligand-binding domain, never before described in literature. A simple polymorphism was excluded through screening of 100 healthy controls. Summary: The patient became pregnant twice (in 2008 and in 2009) and developed PPT after both deliveries. Two months after her first pregnancy and one month after her second pregnancy, she presented with severe endogenous thyrotoxicosis and concomitant suppressed thyrotropin (TSH) levels, which represents an unusual finding in patients affected by RTH. Other causes of hyperthyroidism were excluded. After the hyperthyroid phase, she became hypothyroid (TSH >75 mU/L and low free-thyroxine and free-tri-iodothyronine levels), and eventually returned to her usual euthyroid status. During the course of PPT, no specific treatment was required, except for β-blockers used to treat tachycardia during the hyperthyroid phase. Conclusions: We report a unique case of a woman affected by RTH, due to a novel mutation V283A in THRB, who experienced PPT with a severe thyrotoxic phase after both her pregnancies. The association between RTH and PPT has never been reported in the literature. In particular, the marked suppression of TSH occurring when levels of TH are particularly elevated is not a frequent condition during RTH.
    No preview · Article · Nov 2012 · Thyroid: official journal of the American Thyroid Association