title: Thyroglossal Duct Cyst Carcinomas: A Clinicopathologic Series of 22 Cases with Staging Recommendations.
abstract: Carcinomas arising from thyroglossal duct remnant cysts (TGDCs) are rare, without well-defined management and staging criteria. All TGDCs (n = 685) diagnosed between 2005 and 2015 were retrospectively reviewed, with 22 carcinomas identified (3.2 % incidence). Twenty-two patients (17 females, 5 males), aged 12-64 years (mean 39.9 years; median 39 years) were identified. An anterior, superior midline neck mass was the presenting symptom in all patients. A cancer diagnosis [all papillary thyroid carcinoma (PTC)] was made after the Sistrunk procedure (SP), with a Bethesda Category V or VI classification preoperatively by fine needle aspiration in 5 of 12 cases tested. A SP was performed in all patients, with total thyroidectomy concurrently (n = 4) or subsequently (n = 12). A selected neck dissection was performed in 5 patients, with metastases found in 3. Of the patients who had a thyroidectomy, synchronous PTC was identified in 6 (thus, 6 of 22 patients had synchronous thyroid gland primaries). This supports an origin from extra-thyroidal remnants (cyst origin) rather than metastatic tumor from a thyroid gland primary. Follow-up radioactive iodine therapy was performed in 13 patients. Metastatic disease to local lymph nodes 57 months after presentation was seen in 1 patient, with all others alive and disease free (mean 3.8 years; range 0.4-10.8 years). The TGDCs ranged from 0.8 to 5 cm (mean 2.3 cm), while the PTCs ranged from 0.1 to 3.8 cm (mean 1.4 cm). All of the tumors were classical PTC, showing a sclerotic and infiltrative pattern, with a capsule present in 11. Lymphovascular invasion was detected in 11; margins were positive in 6. Using currently defined criteria, the patients were separated into AJCC stage group I (n = 21) or II (n = 1). However, if extension into the adipose tissue (n = 11), skeletal muscle (n = 10), or perineural/perivascular tissues (n = 10) were used to stage the patients, interpreted to represent the equivalent of "extrathyroidal extension" (n = 13) as defined for thyroid gland primaries, there would be 15 group I and 7 group III cases. All seven group III patients were ≥45 years. Three of four patients with lymph node metastasis also showed soft tissue extension. In conclusion, TGDC carcinomas (TGDCCa) are uncommon, with all classical PTC. For "microcarcinomas" (≤1 cm), conservative management can be used for patients <45 years (i.e., Sistrunk procedure only); for >1 cm tumors, and due to the high incidence of concurrent papillary carcinoma and higher stage at presentation in older patients, completion thyroidectomy is recommended for patients ≥45 years. Thus, even though a good prognosis can be expected for PTC developing in TGDCs, staging is advocated to more appropriately match therapeutic interventions.
authors.affiliation: Southern California Permanente Medical Group, Department of Pathology, Woodland Hills Medical Center, 5601 De Soto Avenue, Woodland Hills, CA, 91365, USA. Lester.D.Thompson@kp.org.
authors.forename: Lester D R
authors.initials: LDR
authors.lastname: Thompson
authors.affiliation: Southern California Permanente Medical Group, Department of Pathology, Woodland Hills Medical Center, 5601 De Soto Avenue, Woodland Hills, CA, 91365, USA.
authors.forename: Hannah B
authors.initials: HB
authors.lastname: Herrera
authors.affiliation: Department of Pathology, Orange County-Anaheim Medical Center, Anaheim, CA, USA.
authors.forename: Sean K
authors.initials: SK
authors.lastname: Lau
mesh_terms: D000293:Adolescent; D000328:Adult; D002277:Carcinoma / Q000473:pathology*; D002291:Carcinoma, Papillary / Q000473:pathology*; D002648:Child; D005260:Female; D006801:Humans; D008297:Male; D008875:Middle Aged; D009367:Neoplasm Staging / Q000379:methods; D009378:Neoplasms, Multiple Primary / Q000473:pathology*; D013955:Thyroglossal Cyst / Q000473:pathology*; D000077273:Thyroid Cancer, Papillary; D013964:Thyroid Neoplasms / Q000473:pathology*; D055815:Young Adult
keywords: Carcinoma, papillary/pathology; Follow-up studies; Incidence; Neck dissection; Prognosis; Thyroglossal cyst/epidemiology; Thyroglossal cyst/pathology; Thyroglossal cyst/surgery; Thyroid neoplasms/pathology; Thyroidectomy