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Hindawi Case Reports in Oncological Medicine Volume 2020, Article ID 6985746, 5 pages https://doi.org/10.1155/2020/6985746 Case Report 1 1 2 Diani Kartini , Sonar S. Panigoro, and Agnes S. Harahap Oncology Division, Department of Surgery, Dr. Cipto Mangunkusumo General Hospital, Faculty of Medicine, Universitas Indonesia, Jakarta, Indonesia Department of Anatomical Pathology, Dr. Cipto Mangunkusumo General Hospital, Faculty of Medicine, Universitas Indonesia, Jakarta, Indonesia Correspondence should be addressed to Diani Kartini; firstname.lastname@example.org Received 18 September 2019; Revised 16 December 2019; Accepted 6 January 2020; Published 16 January 2020 Academic Editor: Peter F. Lenehan Copyright © 2020 Diani Kartini et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. A thyroglossal duct cyst is a lesion that occurs as a result from failure of the thyroglossal duct to obliterate during fetal development. Malignant progression is a rare event that might occur in less than 1% of all cases. Because of its rarity, there are conﬂicting opinions regarding the management of the case. In the present study, a 46-year-old male presented with a painless neck mass that had increased in size over the last 6 months. There was no diﬃculty in swallowing and breathing, change in voice, signiﬁcant weight loss, or any signs of hyperthyroidism. Laboratory workup showed that results were within normal limits. Thyroid gland ultrasonography and cervical contrast CT scan revealed a complex cystic mass that pointed towards a thyroglossal duct cyst. We performed Sistrunk procedure. Postoperative pathology examination revealed microscopic appearance of the thyroglossal duct cyst with a classic follicular variant of papillary thyroid carcinoma. Our latest follow-up showed no signs of tumor recurrence or any complications following surgery on locoregional status. As a ﬁne needle aspiration biopsy cannot ensure a precise result in all of cases, it is essential to perform a solid physical examination and thorough supporting examination in deciding the precise management for the patient. theless, up to one-third of cases may occur in patients aged 20 1. Introduction years and older [8, 9]. Dysphagia, dysphonia, weight loss, or A thyroglossal duct cyst (TGDC) is the most common form rapid growth in size could be signs of malignancy . How- of congenital anomaly in thyroid development . Approxi- ever, this neoplasia is characterized by relatively nonaggres- mately, 70% of midline neck masses in children and 7% of sive property and rarely involves the thyroid gland and locoregional lymph node spread . midline neck masses in adults are thyroglossal duct cysts . Normally, during the third week of fetal development, Sistrunk procedure is a common option chosen for the the thyroid gland descends along the thyroglossal duct, a surgery of a thyroglossal duct cyst, especially for those who structure originating from the foramen caecum of the ton- are categorized into low risk patients, which consists of excision gue, passing through the base of the tongue towards the lower of the thyroglossal duct cyst, the middle part of hyoid bone, and front part of the neck, where it is usually found in adults. The the surrounding tissue around the thyroglossal tract . thyroglossal duct physiologically disappears by the tenth However, in high-risk patients, total thyroidectomy with radio- week of gestation [3, 4]. In some cases, the thyroglossal duct therapy iodine ablation must be considered [12, 13]. Due to the may fail to obliterate and form a thyroglossal duct cyst . rarity of thyroglossal duct cyst carcinoma, there are conﬂicting Malignancy of the thyroglossal duct cyst rarely occurs, only opinions regarding the management approach for this case. in less than 1% of all cases, with papillary carcinoma present- In this report, we present a rare case of papillary carci- ing as the most common type . This malignancy was ﬁrst noma arising in the thyroglossal duct cyst along with a review reported by Brentano in 1911 . An estimate of up to 60% of the literature focusing on the management of malignancy of cases are found in children less than 5 years of age. Never- in the thyroglossal duct cyst. 2 Case Reports in Oncological Medicine Figure 1: Cervical axial CT scan with contrast. Complex cystic mass in the right parasagittal anterior colli area, appears to bulge to the level of the larynx. 2. Case Illustration debris sediment located inside the lesion which raised suspi- cions towards the thyroglossal duct cyst. To obtain a more accurate picture, a CT scan with contrast for the neck area A 46-year-old male came to our surgical oncology clinic pre- senting with a chief complaint of a lump in the anterior com- was performed, which then showed a complex cystic mass partment of the neck, located in the front and slightly to the in the front neck area located in the right parasagittal area, right, which the patient had noticed since a year ago and has sized approximately 3:9×3:8× 5:5 cm, attached to the right been increasing in size in the last 6 months. The patient infrahyoid muscle, appearing to bulge into the larynx, with denied any complaints of diﬃculty in swallowing, diﬃculty diﬀerential diagnosis of the thyroglossal duct cyst or epider- in breathing, change in voice, signiﬁcant weight loss, or any moid cyst (Figure 1). The thyroid gland appeared to be signs of hyperthyroidism. The patient reported to have a his- within normal limits. The scan also discovered multiple tory of dyslipidemia, and during inpatient care, we discov- lymph node enlargement in the submental area and along ered that the patient had hypertension. The patient also the right jugular chain that measured largest in size 22 mm reported to have undergone sinus surgery twice in the last in the right area and 17 mm in the left. The thyroid function 20 years. There was no signiﬁcant family history. test was within normal limits. Physical examination on the patient showed a neck The patient was then diagnosed with a thyroglossal duct mass in the front area, in the midline slightly located to cyst and planned to undergo Sistrunk procedure. The surgery the right, mobile, with soft surface, painless, solid, with a went well, and the patient was discharged the day after. The well-deﬁned border, and sized approximately 5×5×4 cm. excised specimen was then sent to pathology for histopathol- During palpation, no lymph node enlargement was noted. ogy examination. Macroscopic appearance of the specimen Based on the ﬁndings above, the patient was given a working showed a cystic lesion that measured 4×3 × 3 cm with part of the specimen resembling white cauliﬂower and appeared diagnosis of suspected benign right nontoxic goiter (struma nodosa nontoxic). to be fragile (Figure 2). Microscopic examination showed Thyroid gland ultrasonography was performed. The that the cystic lesion consisted of ﬁbrous tissue lined by a sin- result showed that there was no abnormality in the thyroid gle layer of epithelial cells that was erosive in most area. This gland; however, a cystic lesion in the anterior midline area area was the remnant of the thyroglossal duct cyst. Other areas showed that there were tumors in papillary structures was found, sized approximately 3:4×3:5×4:5 cm with Case Reports in Oncological Medicine 3 (a) (b) Figure 2: (a) Unopened excised specimen; (b) opened excised specimen. (a) (b) (c) Figure 3: (a) Duct with a dense connective tissue wall lined with a layer of thorax epithelium that appears to be erosive (HE 40x); (b) tumor tissue with a papillary structure with a ﬁbrovascular stalk (HE 100x); (c) tumor cells with an irregular nucleus with “ground glass nuclei” (blue arrow) and “nuclear grooves” (red arrow) (HE 400x). with a ﬁbrovascular stalk and some in a follicular pattern, lined times accompanied by pain and dysphagia . In our case, by cells that were stratiﬁed, and showing ground glass nuclei, the only signiﬁcant clinical ﬁnding was a painless mass in nuclear grooves, and eosinophilic cytoplasm (Figure 3). The the neck, which increased in size during the last 6 months. histopathology conclusion was a thyroglossal duct cyst with Diﬀerential diagnosis of the thyroglossal duct cyst includes a classic and follicular variant of papillary thyroid carcinoma. a branchial cleft cyst, lipoma, metastasis of thyroid carci- On the 6-month follow-up after surgery, the patient noma, dermoid cyst, sebaceous cyst, and lymph node reported no clinical complaints, and there were no locoregio- enlargement . nal complication nor clinically palpable lymph node enlarge- CT scan, neck MRI, and ultrasonography hold an impor- ment; however, there was a lymph node enhancement tant role in preoperative diagnosis and management plan of discovered by cervical contrast CT scan, and the patient the case , but imaging studies cannot ensure an accurate was planned for a lymph node biopsy; if the biopsy result preoperative diagnosis, and a ﬁne needle aspiration biopsy showed to be malignant, selective neck dissection and total (FNAB) only gives accurate results in 66% of cases. Accord- thyroidectomy would be the treatment of choice. ing to a study by Yang et al., from 17 cases that are reported to have undergone FNAB, results showed 50% to be true pos- itive and 47% to be false negative, mainly due to the hypocel- 3. Discussion lularity of the ﬁrst aspirate and dilution of the cyst . Due to The remnant structure of a thyroglossal duct can exist in the the cystic nature, FNAB is known to yield a low sensitivity and it has been reserved only for investigation of ﬁndings form of a cyst, tract, or duct or as ectopic thyroid tissue located inside the cyst or the duct . The thyroglossal duct suspicious for malignancy such as the presence of calciﬁca- cyst is the most commonly found congenital anomaly of the tions or solid components on ultrasound . Due to the neck in children aged less than 5 years old. An estimate of up low frequency of malignancy on the thyroglossal duct cyst, to 60% of cases is found in children aged less than 5 years old, in the majority of cases, clinicians rarely consider diagnosis of malignancy, which caused FNAB to be rarely performed but almost one-third of cases can appear in patients aged 20 years and older [8, 9]. In our case, this patient belonged in the prior to surgery. However, the possibility of malignancy increases especially in the older population [18, 19]. Further- one-third with an age of 46 years old. Women were a little more, FNAB does not rule out the presence of a malignancy more likely to have this lesion compared to men, with a ratio of 3 : 2 . The most common presenting chief complaint for especially if the clinical suspicion is high . Generally, diagnosis for malignancy of the thyroglossal duct cyst can the thyroglossal duct cyst is asymptomatic neck mass, some- 4 Case Reports in Oncological Medicine There are four approaches regarding surgical manage- be made after the surgery with histopathology examination of the excised specimen . In our case, we performed ment for thyroglossal duct cyst malignancy, which are (1) ultrasonography for the thyroid gland and CT scan for Sistrunk procedure alone , (2) Sistrunk procedure with thyroid lobectomy or pyramidal lobe resection , (3) Sis- the neck area, and the result showed no signs of invasion of the capsule and the surrounding structure and also con- trunk procedure with total or near total thyroidectomy in ﬁrmed no abnormal ﬁndings regarding the thyroid gland. all patients [9, 27], and (4) Sistrunk procedure with selec- A conclusion was drawn from the physical and supporting tive thyroidectomy for high-risk patients [28, 29]. Consid- examination in this case that it was a thyroglossal duct eration to adding thyroid resection in all patients is based on 3 aspects: (1) presence of thyroid malignancy, (2) use cyst with no signs of malignancy. Progression towards malignancy rarely occurs in the of radioactive iodine as adjuvant therapy, and (3) role of thyroglossal duct cyst. The most frequent type of thyroglossal thyroglobulin as a follow-up marker . Sistrunk procedure duct cysts is the papillary type (85%), followed by mixed was introduced in 1920 by Sistrunk which consists of resec- papillary-follicular type (7%), squamous cell type (5%), and tion of the cyst, its tract, the middle part of hyoid bone, and the structure that composes the base of the tongue. By follicular type (1.7%), and also Hurthle cell and anaplastic type (0.9%) which has a worse prognosis [21, 22]. In our case, using this procedure, the recurrence rate could be decreased after the specimen was sent to pathology for histopathology signiﬁcantly compared to simple excision: from 40% (sim- examination, an interesting ﬁnding was brought to light that ple excision) to 1-5% (Sistrunk procedure) [30–32]. Based the type of the tumor did not resemble the most commonly on the study by Balallaa et al., total thyroidectomy is indi- cated without considering the presence of thyroid gland found; rather, it resembled the second most common, which is the mixed papillary-follicular type. involvement clinically or radiologically based on the pre- Management for malignancy of the thyroglossal duct cyst mise that this procedure could assist staging and detect is still widely debated, mainly when the thyroid gland metastasis, and the risk of recurrent laryngeal nerve injury remains in normal condition. Some authors support the the- or parathyroid gland injury is considerably low especially on the hands of an experienced operator . In this case, ory that thyroglossal duct cyst malignancy originated from the ectopic thyroid tissue that is located inside the cyst, while we only performed Sistrunk procedure, considering that some others think that the malignancy occurs from metasta- there was no pathological diagnosis, no thyroid nodule on physical and supporting examination, and no cervical sis of the thyroid gland . The same goes for the surgical management for the case; some authors think the Sistrunk lymph node enlargement noted. procedure to be adequate if there is no abnormality in the thyroid gland. 4. Conclusions Due to rarity of malignancy in the thyroglossal duct cyst, currently, there is no single universal guideline for Malignancy in a thyroglossal duct cyst is a rare event. Physi- the management of the case. Patients are classiﬁed into cal and supporting examinations are both crucial in deciding low risk and high risk according to the revised 2009 how the surgical procedure must be carried out. In our case, American Thyroid Association (ATA) guideline regarding we would continue to observe the locoregional status by both diﬀerentiated thyroid cancer and National Comprehensive physical and supporting examinations. Cancer Network risk stratiﬁcation system with proposed modiﬁcations adjusted to ﬁt for malignancy in the thyro- Conflicts of Interest glossal duct cyst. Patients who are grouped into the low risk category are (1) patients aged between 15 and 45 The authors declare that there are no conﬂict of interest years old, without prior radiation history; (2) tumor size regarding the publication of this article. less than 4 cm; and (3) no distant metastases or lymph node involvement. 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