Papillary thyroid cancer and autoimmune polyglandular syndrome

Abstract

With great interest, we read the article by Karavanaki et al. (1) , who reported on a patient with autoimmune polyglandular syndrome (APS) type II and papillary thyroid carcinoma and highlighted the controversial relationship between thyroid autoimmunity and cancer. We take this opportunity to comment on this debated issue and to describe an overlapping case we have diagnosed and followed recently.In 2005, a 12-year-old boy was admitted at our institution for acute abdominal pain and dehydration; the presence of hyponatremia, hypoglycemia, and hyperkalemia prompted the suspicion of acute mineralocorticoid and glucocorticoid deficiency. Diagnostic tests revealed elevated adrenocorticotropic hormone concentrations ( > 1200 pg/mL) and undetectable serum cortisol levels, confirming a primary adrenal insufficiency of autoimmune origin (serum anti-21-hydroxylase antibody positive titer). Further investigations showed also the coexistence of euthyroid Hashimoto ' s thyroiditis; the boy had positive anti-thyroperoxidase ( > 126 U/mL, normal < 40) and anti-human thyroglobulin (888 U/mL, normal < 35) antibody titer and a mildly enlarged thyroid with decreased echogenicity and inhomogeneous ultrasound pattern. The diagnosis was consistent with APS type II; the patient was prescribed replacement therapy with oral hydrocortisone and fludrocortisone acetate and followed up quarterly. Of note, the recent patient history was positive for an episode of pharyngitis and one of severe gastroenteritis. Results of the laboratory assays were consistent with a previous streptococcal infection (anti-streptolysin O titer > 500 U/mL) and a recent hepatitis A infection (IgM index 3.14, positive > 1). A few months after the diagnosis of APS type II, the thyroid function switched to hypothyroidism with serum thyroid-stimulating hormone (TSH) level of 11.8 mU/L (0.4 -4.4 mU/L) and fT4 level of 9.6 pg/mL (9.8 -16.6 pg/mL); levothyroxine treatment was started. The follow-up of this case lasted until the actual age of 20 years; antibodies against transglutaminase, glutamic acid decarboxylase, tyrosine phosphatase-like molecule, gastric parietal cells, and cholinergic receptor were checked yearly and were persistently negative. Results of the twice-yearly clinical evaluation; levels of vitamin B, folate, calcium, phosphorus, parathormone, and glycemia; platelet counts; and liver enzyme dosages have always been normal, and the boy underwent regular growth and pubertal development. At 16 years of age, his annual thyroid ultrasound, which was also part of the follow-up, revealed a hypoechoic nodule at the right thyroid lobe with a maximum diameter of 15 mm, increased intravascular blood flow, and irregular margins. Fine-needle biopsy cytology was consistent with papillary thyroid carcinoma, and the boy underwent total thyroidectomy and central compartment lymph node dissection. Histology confirmed the cytological diagnosis, and the boy was submitted to radioactive iodine treatment. Currently, serum thyroglobulin levels are undetectable and there is...

Predictors of Malignancy in Children with Thyroid Nodules

“…40,41 In our cohort, we did not detect any role for positive thyroid antibodies; the proportion of differentiated thyroid cancer observed in the negative antibody group and in the autoimmune thyroiditis group overlapped (11% and 12%, respectively), consistent with previous reports on autoimmune thyroiditis in childhood. 14 Although most of the literature reports a predominance of the female sex among cancer patients (1.5:1), 2 in our report, males represent approximately one-half of this group.…”

Co-Existence of Thyroid Nodule and Thyroid Cancer in Children and Adolescents with Hashimoto Thyroiditis: A Single-Center Study

“…Investigation of the relationship between autoimmune thyroid disease and thyroid cancer was started following this study. Some studies suggest that a positive relationship is present between HT and thyroid cancer development, while the reverse is also claimed [9,10,11,12,13,14,15,16]. The number of relevant studies conducted in the childhood period is very low [15,17].…”

Hashimoto's thyroiditis and papillary thyroid cancer: polyglandular hints

“…In all cases in which APS II syndrome was associated with PTC, Hashimoto's thyroiditis was present. Interestingly, this is the third patient of Southern European origin reported (one from Greece and one from Italy already reported); one other patient comes from Brazil [2][3][4].…”

“…Having read the recent article by Ehlers and Schott [1] on the immunological link between Hashimoto's thyroiditis and papillary thyroid cancer (PTC), we would like to provide further insights into this association by presenting the fourth case of autoimmune polyglandular syndrome (APS) II associated with PTC, but the first in which APS syndrome presented with a combination of adrenal insufficiency, autoimmune thyroiditis, and premature ovarian insufficiency (POI) [2][3][4].…”