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Lingual thyroid and hyperthyroidism: A new case and review of the literature

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Abstract

Lingual thyroid is the result of a defective migration of the thyroid anlage occurring between the 3rd and 7th week of gestation. Whereas mutations in the transcription factor-2 (TTF-2) and PAX8 and in the TSH receptor genes (TSH-R) have been reported in a minority of patients with thyroid dysgenesis, the etiopathogeny of the majority of cases, and in particular of thyroid ectopy, remains unclear. The majority of patients with thyroid ectopy are asymptomatic, but obstructive symptoms as well as hypothyroidism have been observed. Hyperthyroidism is an exceptionally rare finding. To our knowledge, only 2 cases have been reported in the literature to date. Herein, we describe an unusual case of thyrotoxicosis related to a nodular lesion in a lingual thyroid. Treatment consisted in restoration of a euthyroid state with thionamide followed by surgical removal of the ectopic gland. The underlying molecular cause of the ectopic lingual thyroid and the toxic adenoma in this case could not be identified. We speculate that abnormally early differentiation of the thyroid gland could interfere with the migration process, a hypothesis yet to be confirmed.

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References

  1. Dossing H., Jorgensen K.E., Oster-Jorgensen E., Krogdahl A., Hegedüs L. Recurrent pregnancy-related upper airway obstruction caused by intratracheal ectopic thyroid tissue. Thyroid 1999, 9: 955–958.

    Article  CAS  PubMed  Google Scholar 

  2. Basaria S., Cooper D. Grave’s disease and recurrent ectopic thyroid tissue. Thyroid 1999, 9: 1261–1264.

    Article  CAS  PubMed  Google Scholar 

  3. Noyek A.M., Friedberg J. Thyroglossal duct and ectopic thyroid disorders. Otolaryngol. Clin. North Am. 1981, 14: 187–201.

    CAS  PubMed  Google Scholar 

  4. Pollice L., Caruso G. Struma cordis. Ectopic thyroid goiter in the right ventricle. Arch. Pathol. Lab. Med. 1986, 110: 452–453.

    CAS  PubMed  Google Scholar 

  5. Roth L.M. Inclusions of non-neoplastic thyroid tissue within cervical lymph nodes. Cancer 1965, 18: 105–111.

    Article  CAS  PubMed  Google Scholar 

  6. Neinas F.W., Gorman C.A., Devine K.D., Woolner L.B. Lingual thyroid: clinical characteristics of 15 cases. Ann. Intern. Med. 1973, 79: 205–210.

    Article  CAS  PubMed  Google Scholar 

  7. Devos H., Rodd C., Gagne N., Laframboise R., Van Vliet G. A search for the possible molecular mechanisms of thyroid dysgenesis: Sex ratios and associated malformations. J. Clin. Endocrinol. Metab. 1999, 84: 2502–2506.

    Article  CAS  PubMed  Google Scholar 

  8. Arancibia P., Veliz J., Barria M., Pineda G. Lingual thyroid: report of three cases. Thyroid 1998, 8: 1055–1057.

    Article  CAS  PubMed  Google Scholar 

  9. Maurice N., Makeieff M., Delol J., et al. La thyroïde ectopique linguale à propos de trois cas. Ann. Otolaryngol. Chir. Cervicofac. 1995, 112: 399–405.

    CAS  PubMed  Google Scholar 

  10. Macchia P.E., Mattei M.G., Lapi P., Fenzi G., Di Lauro R. Cloning, chromosomal localization and identification of polymorphism in the human thyroid transcription factor 2 gene (TITF2). Biochimie 1999, 81: 433–440.

    Article  CAS  PubMed  Google Scholar 

  11. De Felice M., Ovitt C., Biffali E., et al. A mouse model for hereditary thyroid dysgenesis and cleft palate. Nat. Genet. 1998, 19: 395–398.

    Article  PubMed  Google Scholar 

  12. Clifton-Bligh R.J., Wentworth J.M., Heinz P., et al. Mutation of the gene encoding human TTF-2 associated with thyroid agenesis, cleft palate and choanal atresia. Nat. Genet. 1998, 19: 399–401.

    Article  CAS  PubMed  Google Scholar 

  13. Gagne N., Parma J., Deal C., Vassart G., Van Vliet G. Apparent congenital athyreosis contrasting with normal plasma thyroglobulin levels and associated with inactivating mutations in the thyrotropin receptor gene: Are athyreosis and ectopic thyroid distinct entities? J. Clin. Endocrinol. Metab. 1998, 83: 1771–1775.

    CAS  PubMed  Google Scholar 

  14. Abramowicz M.J., Duprez L., Parma J., Vassart G., Heinrichs C. Familial congenital hypothyroidism due to inactivating mutation of the thyrotropin receptor causing profound hypoplasia of the thyroid gland. J. Clin. Invest. 1997, 99: 3018–3024.

    Article  CAS  PubMed Central  PubMed  Google Scholar 

  15. Macchia P.E., Lapi P., Krude H., et al. Pax8 mutations associated with congenital hypothyroidism caused by thyroid dysgenesis. Nat. Genet. 1998, 19: 83–86.

    Article  CAS  PubMed  Google Scholar 

  16. Lapi P., Macchia P.E., Chiovato L., et al. Mutations in the gene encoding thyroid transcription factor -1 (TTF-1) are not a frequent cause of congenital hypothyroidism with thyroid dysgenesis. Thyroid 1997, 7: 383–387.

    Article  CAS  PubMed  Google Scholar 

  17. Perna M.G., Civitareale D., De Filippis V., et al. Absence of mutations in the gene encoding thyroid transcription factor-1 (TTF-1) in patients with thyroid dysgenesis. Thyroid 1997, 7: 377–381.

    Article  CAS  PubMed  Google Scholar 

  18. Koch C.A., Picken C., Clement S.C., Azumi N., Sarlis N.J. Ectopic lingual thyroid: An otolaryngologic emergency beyond childhood. Thyroid 2000, 10: 511–514.

    CAS  PubMed  Google Scholar 

  19. Chanin L.R., Greenberg L.M. Pediatric upper airway obstruction due to ectopic thyroid: classification and case reports. Laryngoscope 1988, 98: 422–427.

    Article  CAS  PubMed  Google Scholar 

  20. Guerin N., Urtasun A., Chauveau E., et al. Thyroïde linguale et kyste thyreo-glosse basi-lingual. A propos de deux cas. Rev. Laryngol. Otol. Rhinol. 1997, 118: 183–188.

    CAS  Google Scholar 

  21. Turgut S., Murat Ozcan K., Celikkanat S., Katirci H., Ozdem C. Diagnostic et traitement de la thyroïde linguale: revue de la littérature. Rev. Laryngol. Otol. Rhinol. 1997, 118: 189–192.

    CAS  Google Scholar 

  22. Kamat M.R., Kulkarni J.R., Desai P.B., Jussawalla D.J. Lingual thyroid: A review of 12 cases. Br. J. Surg. 1979, 66: 537–539.

    Article  CAS  PubMed  Google Scholar 

  23. Kaplan M., Kauli R., Lubin E., Grunebaum M., Laron Z. Ectopic thyroid gland. A clinical study of 30 children and review. J. Pediatr. 1978, 92: 205–209.

    Article  CAS  PubMed  Google Scholar 

  24. Kuehn P.G., Newell R.C., Reed J.F. Exophtalmos in a woman with lingual, subhyoid and lateral lobe thyroid glands. N. Engl. J. Med. 1966, 274: 652–654.

    Article  CAS  PubMed  Google Scholar 

  25. Azzam A., Nitunga N., Baulieu J.L., et al. Ectopie thyroïdienne et maladie de Basedow. Presse Med. 1988, 17: 383–385.

    CAS  PubMed  Google Scholar 

  26. Simkin P.H., Ramierz L.A., Zweizig S.L., et al. Monomorphic teratoma of the ovary: a rare cause of triiodothyronine toxicosis. Thyroid 1999, 9: 949–954.

    Article  CAS  PubMed  Google Scholar 

  27. Grüters A., Schöneberg T., Biebermann H., et al. Clinical Case Seminar: Severe congenital hyperthyroidism caused by a germ-line neo mutation in the extracellular portion of the thyrotropin receptor. J. Clin. Endocrinol. Metab. 1998, 83: 1431–1436.

    PubMed  Google Scholar 

  28. Duprez L., Parma J., Van Sande J., et al. Germline mutations in the thyrotropin receptor gene cause non-autoimmune autosomal dominant hyperthyroidism. Nat. Genet. 1994, 7: 396–401.

    Article  CAS  PubMed  Google Scholar 

  29. Tonacchera M., Van Sande J., Cetani F., et al. Functional characteristics of three new germline mutations of the thyrotropin receptor gene causing autosomal dominant toxic thyroid hyperplasia. J. Clin. Endocrinol. Metab. 1996, 81: 547–554.

    CAS  PubMed  Google Scholar 

  30. Kopp P., Jameson L., Roe T. Congenital nonautoimmune hyperthyroidism in a nonidentical twin caused by a sporadic germline mutation in the thyrotropin receptor gene. Thyroid 1997, 7: 765–770.

    Article  CAS  PubMed  Google Scholar 

  31. Esapa C.T., Duprez L., Ludgate M., et al. A novel thyrotropin receptor mutation in a infant with severe thyrotoxicosis. Thyroid 1999, 9: 1005–1010.

    Article  CAS  PubMed  Google Scholar 

  32. Van Sande J., Parma J., Tonacchera M., et al. Somatic and germline mutations of the TSH receptor gene in thyroid diseases. J. Clin. Endocrinol. Metab. 1995, 80: 2577–2585.

    PubMed  Google Scholar 

  33. Führer D., Kubisch C., Scheibler U., et al. The extracellular thyrotropin receptor domain is not a major candidate for mutations in toxic thyroid nodules. Thyroid 1998, 8: 997–1001.

    Article  PubMed  Google Scholar 

  34. Duprez L., Parma J., Van Sande J., et al. Pathology of the TSH receptor. J. Pediatr. Endocrinol. Metab. 1999, 12: 295–302.

    PubMed  Google Scholar 

  35. Parma J., Van Sande J., Swillens S., et al. Somatic mutations causing constitutive activity of the thyrotropin receptor are the major cause of hyperfunctioning thyroid adenomas: identification of additional mutations activating both the cyclic adenosine 3′,5′-monophosphate and inositol phosphate-Ca2+ cascades. Mol. Endocrinol. 1995, 9: 725–733.

    CAS  PubMed  Google Scholar 

  36. Takeshita M., Nagayama Y., Yokoyama N., et al. Rarity of oncogenic mutations in the thyrotropin receptor of autonomously functioning thyroid nodules in Japan. J. Clin. Endocrinol. Metab. 1995, 80: 2607–2611.

    CAS  PubMed  Google Scholar 

  37. Biebermann H., Liesenkotter K.P., Emeis M., Oblanden M., Grüters A. Severe congenital hypothyroidism due to a homozygous mutation of the beta TSH gene. Pediatr. Res. 1999, 46: 170–173.

    Article  CAS  PubMed  Google Scholar 

  38. Doeker B.M., Pfaffle R.W., Pohlenz J., Andler W. Congenital central hypothyroidism due to a homozygous mutation in the thyrotropin beta-subunit gene follows an autosomal recessive inheritance. J. Clin. Endocrinol. Metab. 1998, 83: 1762–1765.

    CAS  PubMed  Google Scholar 

  39. Medeiros-Neto G., Herodotou D.T., Rajan S., et al. A circulating, biologically inactive thyrotropin caused by a mutation in the beta subunit gene. J. Clin. Invest. 1996, 97: 1250–1256.

    Article  CAS  PubMed Central  PubMed  Google Scholar 

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Correspondence to P. Caron.

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Abdallah-Matta, M.P., Dubarry, P.H., Pessey, J.J. et al. Lingual thyroid and hyperthyroidism: A new case and review of the literature. J Endocrinol Invest 25, 264–267 (2002). https://doi.org/10.1007/BF03344002

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