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Journal of Clinical Medicine Oct 2023Antithyroid drug-induced agranulocytosis (AIA) (neutrophils <500/µL) is a rare but serious complication in the treatment of hyperthyroidism.
BACKGROUND
Antithyroid drug-induced agranulocytosis (AIA) (neutrophils <500/µL) is a rare but serious complication in the treatment of hyperthyroidism.
METHODOLOGY
Adult patients with AIA who were followed up at 12 hospitals in Spain were retrospectively studied. A total of 29 patients were studied. The etiology of hyperthyroidism was distributed as follows: Graves' disease ( = 21), amiodarone-induced thyrotoxicosis ( = 7), and hyperfunctioning multinodular goiter ( = 1). Twenty-one patients were treated with methimazole, as well as six patients with carbimazole and two patients with propylthiouracil.
RESULTS
The median (IQR) time to development of agranulocytosis was 6.0 (4.0-11.5) weeks. The most common presenting sign was fever accompanied by odynophagia. All of the patients required admission, reverse isolation, and broad-spectrum antibiotics; moreover, G-CSF was administered to 26 patients (89.7%). Twenty-one patients received definitive treatment, thirteen patients received surgery, nine patients received radioiodine, and one of the patients required both treatments. Spontaneous normalization of thyroid hormone values occurred in six patients (four patients with amiodarone-induced thyrotoxicosis and two patients with Graves' disease), and two patients died of septic shock secondary to AIA.
CONCLUSIONS
AIA is a potentially lethal complication that usually appears around 6 weeks after the initiation of antithyroid therapy. Multiple drugs are required to control hyperthyroidism before definitive treatment; additionally, in a significant percentage of patients (mainly in those treated with amiodarone), hyperthyroidism resolved spontaneously.
PubMed: 37892693
DOI: 10.3390/jcm12206556 -
Turkish Archives of Otorhinolaryngology Sep 2023The aim of the study was to retrospectively analyze the patients who underwent thyroidectomy at a tertiary academic center regarding their surgical indications,...
OBJECTIVE
The aim of the study was to retrospectively analyze the patients who underwent thyroidectomy at a tertiary academic center regarding their surgical indications, histopathological diagnosis, and surgical complications.
METHODS
The study included a total of 739 consecutive patients who underwent lobectomy, total thyroidectomy, or completion thyroidectomy performed under intraoperative nerve monitoring (IONM) at the Department of Otorhinolaryngology-Head and Neck Surgery of Dokuz Eylül University between January 2009 and December 2019. Demographic data of the patients, preoperative clinicopathological characteristics, postoperative complications, characteristics of surgery and histopathological results were evaluated.
RESULTS
There were 619 patients in the primary surgery and 120 patients in the revision surgery groups. Indications for surgery were suspicion of malignancy in 486, multinodular goiter in 214, and hyperthyroidism in 39 patients. Final histopathological evaluation of specimens revealed malignancy in a total of 507 (68.6%) patients. Rates of transient and permanent hypocalcemia were 7.3% (54/739) and 2.2% (16/739) in the whole group, while this was 6.6% (41/619) and 1.5% (9/619), respectively, among primary total thyroidectomy patients. There were 61 (8.3%) patients with transient recurrent laryngeal nerve (RLN) paralysis (unilateral in 60 patients, bilateral in one patient) and five (0.7%) patients with permanent unilateral RLN paralysis as postoperative complications. Rates for postoperative hematoma, seroma, wound infection and chylous fistula were 2.2%, 3.7%, 0.1%, and 0.5%, respectively.
CONCLUSION
Our results support the safety of thyroid surgery performed under IONM in tertiary academic centers. Every institution should document and share its own results to properly inform its patients preoperatively.
PubMed: 38020413
DOI: 10.4274/tao.2023.2023-5-19 -
Clinical Case Reports Dec 2023Prompt coordinated care enables vaginal delivery for severe goiter-induced airway compression in late pregnancy when expectant management is precluded.
KEY CLINICAL MESSAGE
Prompt coordinated care enables vaginal delivery for severe goiter-induced airway compression in late pregnancy when expectant management is precluded.
ABSTRACT
Long-standing neglected multinodular goiter rarely causes life-threatening airway compression in late pregnancy. Most cases report cesarean delivery to relieve respiratory distress. Vaginal birth may also be possible, but the evidence is limited. A 30-year-old woman with a 7-year goiter history developed severe dyspnea at 34 weeks gestation. Despite the risks, labor was induced by urgent decompression. She vaginally delivered a premature but healthy infant. Her breathing improved after delivery. She later underwent an uncomplicated thyroidectomy. With careful selection and monitoring, vaginal delivery can be considered for goiter-induced respiratory compromise when expectant management is precluded. This case demonstrates successful urgent decompression and favorable maternal-fetal outcomes are possible with a coordinated approach despite the high-risk scenario.
PubMed: 38084351
DOI: 10.1002/ccr3.8303 -
Familial Cancer Oct 2023Pathogenic germline DICER1 variants are associated with pleuropulmonary blastoma, multinodular goiter, embryonal rhabdomyosarcoma and other tumour types, while mosaic...
Pathogenic germline DICER1 variants are associated with pleuropulmonary blastoma, multinodular goiter, embryonal rhabdomyosarcoma and other tumour types, while mosaic missense DICER1 variants in the RNase IIIb domain are linked to cause GLOW (global developmental delay, lung cysts, overgrowth, and Wilms' tumor) syndrome. Here, we report four families with germline DICER1 pathogenic variants in which one member in each family had a more complex phenotype, including skeletal findings, facial dysmorphism and developmental abnormalities. The developmental features occur with a variable expressivity and incomplete penetrance as also described for the neoplastic and dysplastic lesions associated with DICER1 variants. Whole exome sequencing (WES) was performed on all four cases and revealed no further pathogenic or likely pathogenic dominant, homozygous or compound heterozygous variants in three of them. Notably, a frameshift variant in ARID1B was detected in one patient explaining part of her phenotype. This series of patients shows that pathogenic DICER1 variants may be associated with a broader phenotypic spectrum than initially assumed, including predisposition to different tumours, skeletal findings, dysmorphism and developmental abnormalities, but genetic work up in syndromic patients should be comprehensive in order not to miss additional underlying /modifying causes.
Topics: Female; Humans; Germ-Line Mutation; Phenotype; Frameshift Mutation; Cysts; Ribonuclease III; Germ Cells; DEAD-box RNA Helicases
PubMed: 34331184
DOI: 10.1007/s10689-021-00271-z -
Endocrine Connections Dec 2023An improvement in iodine status in Veneto Region has been documented in the last decade. We aimed at estimating the incidence of hyperthyroidism in the Veneto Region...
OBJECTIVE
An improvement in iodine status in Veneto Region has been documented in the last decade. We aimed at estimating the incidence of hyperthyroidism in the Veneto Region (Italy) over the period 2013-2022.
METHODS
Retrospective population-based study conducted in Veneto (4.9 million people) using the population registry, an administrative health database. Between 2012 and 2022, hyperthyroidism incidence was defined thank to a health-care co-payment exemption for hyperthyroidism or any hospital diagnosis of hyperthyroidism. Incident hyperthyroidism was defined from 2013 to 2022 to exclude prevalent cases. Standardized incidence rates (IRs) were reported by age, sex, and etiology of thyroid hyperfunction too.
RESULTS
We identified 26,602 incident cases (IR of 54.38 per 100,000 person-years, 2.47-fold higher in females than in males). IR decreased from 69.87 (95% CI: 67.49, 72.25) in 2013 to 42.83 (95% CI: 40.99, 44.66) in 2022. In 2020, an out-of-trend decrease in hyperthyroidism incidence was documented, corresponding to the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) pandemic outbreak, with a realignment to the trend in the subsequent years. The annual percentage change according to the cause of hyperthyroidism was as follows: -6.62% (95% CI: 8.47, 4.73) (P < 0.0001) in toxic multinodular goiter, -7.56% in toxic uninodular goiter (95% CI: 10.54, 4.48) (P < 0.001) and -4.70% (95% CI: 6.33, 3.04) in toxic diffuse goiter (Graves' disease) (P < 0.001).
CONCLUSIONS
We documented a decline in the incidence of hyperthyroidism in Veneto Region, paralleling the improvement of the iodine status, thanks to a long and sustained iodine prophylaxis campaign. SARS-CoV-2 pandemic and vaccination campaign did not change the declining trend of hyperthyroidism incidence in our study region.
SIGNIFICANCE STATEMENT
An improvement in iodine status in the population residing in the Veneto region has been documented in the last decade, thanks to a nationwide voluntary iodine prophylaxis program running since 2005, but its impact on the epidemiology of thyroid disease has never been documented. This is the largest study on the incidence rates of hyperthyroidism carried out in Italy and covers the longest observation period among all regionwide population-based studies of hyperthyroidism in our country. We documented a reduction in the incidence of hyperthyroidism, which was more pronounced in nodular goiter diagnosis but involved also toxic diffuse goiter. The decline in the incidence of hyperthyroidism in Veneto Region shows the efficacy and safety of the iodine prophylaxis campaign.
PubMed: 37855396
DOI: 10.1530/EC-23-0292 -
Archives of Endocrinology and Metabolism Feb 2024Mutations in DICER1 are found in differentiated thyroid carcinoma (DTC) and in multinodular goiter (MNG) at a younger age with other tumors, which characterizes DICER1...
OBJECTIVE
Mutations in DICER1 are found in differentiated thyroid carcinoma (DTC) and in multinodular goiter (MNG) at a younger age with other tumors, which characterizes DICER1 syndrome. DICER1 is one driver to DTC; however, it is also found in benign nodules. We speculated that patients with mutations in DICER1 may present long-lasting MNG. Our aim was to investigate the frequency of DICER1 variants in patients with MNG.
SUBJECTS AND METHODS
Patients who submitted to total thyroidectomy due to large MNG with symptoms were evaluated. DICER1 hotspots were sequenced from thyroid nodule samples. To confirm somatic mutation, DNA from peripheral blood was also analyzed.
RESULTS
Among 715 patients, 154 were evaluated with 56.2 ± 12.3 years old (28-79) and the thyroid volume was 115.7 ± 108 mL (16.2-730). We found 11% with six DICER1 variations in a homo or heterozygous state. Only rs12018992 was a somatic DICER1 variant. All remaining variants were synonymous and likely benign, according to the ClinVar database. The rs12018992 was previously described in an adolescent with DTC, measuring 13 mm. There were no significant differences according to gender, familial history of goiter, age, thyroid volume, TSH and TI-RADS classification between DICER1 carriers. Free T4 were lower in patients with DICER1 polymorphisms (13.77 ± 1.8 vs. 15.44 ± 2.4 pmol/L, p = 0.008), regardless of TSH levels.
CONCLUSION
We conclude that germline DICER1 variants can be found in 11% of large goiters but no second-hit somatic mutation was found. DICER1 is one driver to thyroid lesion and a second-hit event seems unnecessary in the MNG development.
Topics: Adolescent; Adult; Aged; Humans; Middle Aged; Adenocarcinoma; DEAD-box RNA Helicases; Goiter, Nodular; Prevalence; Ribonuclease III; Thyroid Neoplasms; Thyrotropin
PubMed: 38330293
DOI: 10.20945/2359-4292-2023-0030 -
Problemy Endokrinologii Oct 2023DICER1 syndrome is a rare genetic disorder with the progressive development of malignant and non-malignant diseases in childhood. The cause of this syndrome is a... (Review)
Review
DICER1 syndrome is a rare genetic disorder with the progressive development of malignant and non-malignant diseases in childhood. The cause of this syndrome is a dusfunction of the endoribonuclease DICER, which plays an important role in the processing of microRNAs with subsequent regulation of the control of the expression of oncogenes and tumor suppressor genes. Clinical manifestations of dyseropathies is very different and may include both endocrine manifestations - multinodular goiter, differentiated thyroid cancers, ovarian stromal tumors, pituitary blastoma, and non-endocrine formations - pleuropulmonary blastoma, cystic nephroma, pineoblastoma. The presence of somatic mutations of the DICER1 gene is a resultant stage in the pathogenesis of dyseropathies, determining the further path of oncogenesis. At present, DICER1 syndrome is diagnosed extremely rarely, which leads to late detection of the components of the disease in the patient, late diagnosis of neoplasms, lack of family counseling. Diagnosis at the early stages of the disease, the development of screening programs for the management of these patients allows minimizing the risks of developing more malignant, aggressive forms of the disease.
Topics: Humans; Ribonuclease III; DEAD-box RNA Helicases; Mutation; Female; Thyroid Neoplasms; Goiter, Nodular; Pulmonary Blastoma
PubMed: 38796764
DOI: 10.14341/probl13383 -
Cureus Apr 2024Total thyroidectomy is evolving as the choice of treatment for non-malignant thyroid conditions. Therefore, an ideal method of thyroxine replacement is necessary to...
INTRODUCTION
Total thyroidectomy is evolving as the choice of treatment for non-malignant thyroid conditions. Therefore, an ideal method of thyroxine replacement is necessary to avoid the ill effects of under- and over-replacement in such patients.
AIM
To assess the correlation between optimal thyroxine dose and potential variables like lean body mass (LBM), body surface area (BSA), body mass index (BMI), body weight, age, and sex in patients who underwent total thyroidectomies for benign multinodular goiters in our institute.
MATERIALS AND METHODS
A longitudinal cohort study was undertaken at the Government Medical College Thrissur, a tertiary care provider in India, between October 2018 and September 2019. One hundred adult patients who underwent a total thyroidectomy for various benign thyroid conditions were included. They were initially given thyroxine 75 µg upon discharge and received follow-up doses every two months until they achieved euthyroid status on two consecutive visits. The variables evaluated at this stage included age, sex, actual body weight, lean body weight, BMI, and biochemical data (triiodothyronine (T3), thyroxine (T4), thyroid-stimulating hormone (TSH)). Correlation, multiple step-wise regression, and variance were carried out using EPI INFO version 7.2.2.6.
RESULTS
The best predictors for optimum thyroxine dose were BSA (0.923, P < 0.01) and LBM (0.921, P < 0.01), compared with body weight (0.833, P < 0.01) and BMI (0.523, P < 0.01). In our study, the least significant factor was the age of the patient (r = 0.117, P < 0.01). There was no significant association between gender and thyroxine dose. The mean thyroxine dose was 1.87 µg/kg of the patient's body weight.
CONCLUSION
The optimum thyroxine replacement based on BSA or LBM is a more ideal method than based on BMI or body weight alone.
PubMed: 38765445
DOI: 10.7759/cureus.58430 -
Surgical Endoscopy Dec 2023Postoperative hypocalcemia is a common complication of thyroidectomy. This problem is most often associated with accidental devascularization or excision of the...
BACKGROUND
Postoperative hypocalcemia is a common complication of thyroidectomy. This problem is most often associated with accidental devascularization or excision of the parathyroid glands (PG).
AIM
Aim was to study near-infrared (NIR) fluorescent imaging with intraoperative PG indocyanine green (ICG) angiography to help identify and preserve PG during total thyroidectomy in order to avoid postoperative hypocalcemia.
MATERIAL AND METHODS
From 2017 to 2022, a total of 92 patients underwent total thyroidectomy at Odessa Regional Hospital. Indications for surgery were multinodular goiter (n = 42), thyroid cancer (n = 43), and Graves' disease (n = 7). By randomization all patients were divided into two groups: in the control group, 48 patients underwent standard total thyroidectomy, and in the main group, 44 patients underwent NIR-assisted total thyroidectomy with ICG angiography. Serum calcium and parathyroid hormone levels were compared between the two groups of patients in 1, 7-15 days after surgery and then 3, 6 months later.
RESULTS
In the control group, based on a visual assessment of the PG, autotransplantation of the PG was conducted in only five cases. In the second group, autotransplantation was performed in 16 patients. The transient postoperative hypocalcemia was observed in 8 patients of the control group (16, 70%) and in the 2 patients of ICG group (4, 50%) on 5-10 postoperative days. In the first group, 2 patients at 3 months after surgery had permanent hypocalcaemia.
CONCLUSION
NIR fluorescent imaging with intraoperative PG ICG angiography is a safe and an easily repeatable method. This technique provides improved detecting and assessment of the perfusion of the PG. The need for autotransplantation of the PG can be determined more objectively using ICG imaging than simple visualization.
Topics: Humans; Indocyanine Green; Hypocalcemia; Hypoparathyroidism; Parathyroid Glands; Angiography; Thyroidectomy; Coloring Agents; Postoperative Complications; Parathyroid Hormone
PubMed: 37721589
DOI: 10.1007/s00464-023-10466-3 -
International Journal of Surgery Case... Aug 2023In areas with iodine deficiency, multinodular goiter is a prevalent cause of hyperthyroidism. If left untreated, it can grow until it is noticeably huge in size....
INTRODUCTION
In areas with iodine deficiency, multinodular goiter is a prevalent cause of hyperthyroidism. If left untreated, it can grow until it is noticeably huge in size. Although it is a rare clinical phenomenon, thyroid swelling extending into the retro-pharyngeal space presents a substantial difficulty for the surgeon. In this article, we report a giant thyroid mass that had extended to the retro-pharyngeal space and how it posed a challenge during surgery.
CASE PRESENTATION
Female, 30-year-old, presented with a huge goiter of 15 years duration which extended to retro-visceral space and completely encircled them without extension to retro-sternal space. After the patient was rendered euthyroid with propylthiouracil (PTU), the thyroid mass was removed surgically. Postoperatively, the patient developed only a transient, asymptomatic hypocalcemia, as a complication.
DISCUSSION
If a goiter is large enough, it can grow inferiorly into the mediastinum and outside the typical boundaries of the thyroid bed. However, they rarely grow into the retro-pharyngeal space and present a management issue. The management of this patient was also more complicated as the goiter grew enormous and giant, particularly when it came to controlling the airway and surgically removing the tumor.
CONCLUSION
A preoperative CT scan is an important part of diagnostic studies for a giant goiter to diagnose extrathyroidal bed extension. Total or near-total thyroidectomy through a large cervical incision is the mainstay of treatment for such patients to relieve compression symptoms and treat associated hyperthyroidism.
PubMed: 37487349
DOI: 10.1016/j.ijscr.2023.108543