Parathyroid adenoma with prominent lymphocytic infiltrate having histological features highly suggestive of IgG4-related disease: a case report and literature review

Abstract

Parathyroid adenoma with prominent lymphocytic infiltrate is a rare disease. Until now, 11 patients have been reported. Herein, we report a 57-year-old man who had a neck mass that was incidentally found. Aspiration cytology and subsequent needle biopsy of the tumor were performed and suggested papillary thyroid carcinoma. From the resected specimen, however, the patient was finally diagnosed with parathyroid adenoma with prominent lymphocytic infiltrate, characterized by hyperplastic parathyroid cells with nuclear atypia within fibrotic stroma along with numerous lymphocytes forming germinal centers. Some eosinophils and plasma cells were also observed with some histological features highly suggestive of IgG4-related disease (IgG4-RD), including increased IgG4-positive plasma cells and IgG4/IgG-positive plasma cell ratio, storiform-type fibrosis, and obliterative phlebitis. It turned out that microfollicular or trabecular architecture and cellular atypia with high expression of HBME-1 observed in the aspiration cytology and needle biopsy had been misinterpreted as a thyroid malignancy. This is the first report describing microscopic features of aspiration cytology and needle biopsy of parathyroid adenoma with prominent lymphocytic infiltrate, warning that it can mimic papillary thyroid carcinoma in biopsy specimens. Furthermore, the IgG4-RD-like features of the present case and previous reports imply that parathyroid adenoma with prominent lymphocytic infiltrate may be a type of IgG4-RD.

IgG4‐related disease presenting as severely symptomatic hypercalcemia: A case report and review of literature

“…Primary hyperparathyroidism with elevated serum PTH levels were found in 5 cases 12–14,16,17 . Among them, 2 patients 13,14 were found to have parathyroid adenomas on imaging and pathological specimens, while the other 2 patients 12,16 had normal parathyroid imaging examinations, and the 5th patient 17 did not have parathyroid imaging examinations.…”

“…7,10 Hypercalcemia occurring in patients with IgG4-RD is not common. A literature review showed that only 5 definitely [11][12][13][14][15] (including 1 conference abstract 15 ) and 2 possible (not biopsied) IgG4-RD 16,17 associated hypercalcemia had been reported (Table 1). According to the literature, the pathogenesis of IgG4-RD-associated hypercalcemia can be divided into parathyroid hormone (PTH)-related and non-PTH-related.…”

“…Primary hyperparathyroidism with elevated serum PTH levels were found in 5 cases. [12][13][14]16,17 Among them, 2 patients 13,14 were found to have parathyroid adenomas on imaging and pathological specimens, while the other 2 patients 12,16 had normal parathyroid imaging examinations, and the 5th patient 17 did not have parathyroid imaging examinations. Jung et al 13 reported a 57-year-old man was incidentally found with a well-demarcated mass in the left thyroid, and histopathological and immunohistochemical (IHC) findings led to the diagnosis of parathyroid adenoma with prominent lymphocytic infiltrate having features consistent with the diagnosis of IgG4-RD, which suggested IgG4-RD-related isolated parathyroid involvement.…”

“…[12][13][14]16,17 Among them, 2 patients 13,14 were found to have parathyroid adenomas on imaging and pathological specimens, while the other 2 patients 12,16 had normal parathyroid imaging examinations, and the 5th patient 17 did not have parathyroid imaging examinations. Jung et al 13 reported a 57-year-old man was incidentally found with a well-demarcated mass in the left thyroid, and histopathological and immunohistochemical (IHC) findings led to the diagnosis of parathyroid adenoma with prominent lymphocytic infiltrate having features consistent with the diagnosis of IgG4-RD, which suggested IgG4-RD-related isolated parathyroid involvement. Üsküdar Cansu et al's report 14 showed that although their patient was found having parathyroid adenoma on imaging and surgical specimen, histopathological findings showed that lymphoplasmacytic cells were noted in the adipose tissue around the parathyroid gland, but the parathyroid gland and adenoma were free of infiltration, and hypercalcemia of the patient was unresponsive to high-dose corticosteroid treatment.…”

Sağlık Bilimlerinde Akademik Analiz ve Tartışmalar

“…However, powdered formulas are not sterile products because of their current production processes and may contain pathogenic microorganisms (Kent 2015). Infants are highly susceptible to foodborne infections due to their immature immune and gastrointestinal systems, and even possible microbial contamination in infant formula can cause serious illness and even death (Cho 2019;Zheng 2021). In terms of baby health and survivability, it is of critical importance that infant formulas meet national and international criteria and have high microbiological quality (Kent 2015).…”