Skip to main content

Advertisement

SpringerLink
Log in

Bilobar thyroid agenesis with primary hyperparathyroidism: report of a case

  • Case Report
  • Published:
Surgery Today Aims and scope Submit manuscript

Abstract

Congenital thyroid abnormalities are rarely seen. They mostly include hemiagenesis with or without involving the isthmus. In this report, we present a case of bilateral lobe agenesis with hypertrophied isthmus and high calcium and elevated PTH levels which were detected during routine examinations of a 49-year-old female patient. Some findings consistent with parathyroid pathology on the right side were determined in parathyroid scintigraphy. At thyroid scan and neck ultrasonography there was no sign of bilateral thyroid tissue except a mass localized isthmus. The right lower parathyroidectomy and thyroidectomy for isthmus were performed; the pathology report was shown as parathyroid adenoma and nodular colloidal goiter. This case of bilobar agenesis with incidental primary hyperparathyroidism due to single parathyroid adenoma is the first case reported in literature.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5

References

  1. Leatherdale BA. An unusual thyroid gland. Br J Surg. 1973;60:410–3.

    Article  CAS  PubMed  Google Scholar 

  2. Handfield-Jones. In: Henle J, ed. Handbuch der Systematischen Anatomie des Menschen. Sohn: Friedrich Vlewig und Braunschweig; 1866.p. 538.

  3. Sakurai K, Amano S, Enomoto K, Matsuo S, Kitajima A. Primary hyperparathyroidism with thyroid hemiagenesis. Asian J Surg. 2007;30(2):151–3.

    Article  PubMed  Google Scholar 

  4. Zhao S, Chen X, Zhu G. Infant’s bilobar thyroid agenesis and mother’s I-131 accidental administration. Ann Endocrinol (Paris). 2013;74(1):62–4.

    Article  PubMed  Google Scholar 

  5. Gaudino R, Garel C, Czernichow P, Léger J. Proportion of various types of thyroid disorders among newborns with congenital hypothyroidism and normally located gland: a regional cohort study. Clin Endocrinol (Oxf). 2005;62:444–8.

    Article  PubMed  Google Scholar 

  6. Tonacchera M, Banco ME, Montanelli L, Di Cosmo C, Agretti P, De Marco G, et al. Genetic analysis of the PAX8 gene in children with congenital hypothyroidism and dysgenetic or ectopic thyroid glands: identify action of a novel sequence variant. Clin Endocrinol (Oxf). 2007;67:34–40.

    Article  CAS  PubMed  Google Scholar 

  7. Mydlarz WK, Zhang K, Micchelli ST, Kim M, Tufano RP. Ipsilateral double parathyroid adenoma and thyroid hemiagenesis. ORL J Otorhinolaryngol Relat Spec. 2010;72:272–4.

    Article  CAS  PubMed  Google Scholar 

  8. Isreb S, Alem F, Smith D. Left thyroid hemiagenesis in a patient with primary hyperthyroidism. BMJ Case rep. 2010. doi:10.1136/bcr.03.2010.2864.

    Google Scholar 

  9. Aslaner A, Aydin M, Ozdere A. Multinodular goiter with thyroid hemiagenesis: a case report and review of the literature. Acta Chir Belg. 2005;105:528–30.

    CAS  PubMed  Google Scholar 

  10. Karabay N, Comlekci A, Canda MS, Bayraktar F, Degirmenci B. Thyroid hemiagenesis with multinodular goiter: a case report and review of the literature. Endocr J. 2003;50:409–13.

    Article  PubMed  Google Scholar 

  11. Melnick JC, Stemkowski PE. Thyroid hemiagenesis (hockey stick sign): a review of world literature and a report of 4 cases. J Clin Endocrinol Metab. 1981;52:247–51.

    Article  CAS  PubMed  Google Scholar 

  12. Tiwari PK, Baxi N, Baxi J, Koirala D. Right-sided hemiagenesis of the thyroid lobe and isthmus: a case report. Indian J Radiol Imaging. 2008;18(4):313–5.

    Article  PubMed Central  PubMed  Google Scholar 

  13. Vázquez-Chávez C, Acevedo-Rivera K, Sartorius C, Espinosa-Said L. Thyroid hemiagenesis: report of 3 cases and review of the literature. Gac Med Mex. 1989;125:395–9.

    PubMed  Google Scholar 

  14. Schanaider A, de Oliveira PJ. Thyroid isthmus agenesis associated with solitary nodule: a case report. Cases J. 2008;1(1):211.

    Article  PubMed Central  PubMed  Google Scholar 

  15. Dixit D, Shilpa MB, Harsh MP, Ravishankar MV. Agenesis of isthmus of thyroid gland in adult human cadavers: a case series. Cases J. 2009;2:6640.

    Article  PubMed Central  PubMed  Google Scholar 

  16. Maiorana R, Carta A, Floriddia D, Leonardi D, Buscema M, Sava L, et al. Thyroid hemiagenesis: prevalence in normal children and effect on thyroid function. JCEM. 2003;88:1534–2153.

    CAS  PubMed  Google Scholar 

  17. Ranade AV, Rai R, Pai MM, Nayak SR, Prakash, Krisnamurthy A, et al. Anatomic variations of the thyroid gland: possible surgical implication. Singap Med J. 2008;49:831.

    CAS  Google Scholar 

  18. Kumar TR, Pradeep PV, Ragavan M. Bilobar thyroid agenesis presenting with adenomatous isthmus and hypothyroidism in a 13 year old girl: a case report. J Ped Sci. 2010;2(2):e21.

    Google Scholar 

  19. Attipou K, Cheynel N, Aubry K, Pech de Laclause B, Durand-Fontainer S, Vallaix D, et al. Bilobar thyroid agenesis. Ann Endocrinol. 2000;61:509–10.

    CAS  Google Scholar 

  20. Dutta M, Ghatak S, Saha J, Sinha R. Bilobar agenesis of thyroid gland with colloidal change of the isthmus and subclinical hypothyroidism: a case report. Int J Ped Otorhinolaryngol Extra. 2011;6:243–5.

    Article  Google Scholar 

  21. Sharma P, Karki B, Ghimire PG, Shrestha M, Ghimire A, Pathak A. Bilobar agenesis of thyroid gland with hypertrophied isthmus. J Inst Med. 2010; 32:251–2.

  22. Gauger PG. Thyroid gland In: Mulholland MW, Lillemoe KD, editors. Greenfield’s Surgery: Scientific principles and practice editors. 4th ed Lippincott Williams & Wilkins, Philadelphia, 2006, p. 1–62.

  23. Merida-Velasco JA, Garcia-Garcia JD, Espin-Ferra J, Linares J. Origin of the ultimobranchial body and its colonizing cells in human embryos. Acta Anat. 1989;136:325–30.

    Article  CAS  PubMed  Google Scholar 

  24. Pintar JE. Normal development of the hypothalamic-pituitary–thyroid axis. In: Braverman LE, Utiger RD, editors. Werner and Ingmar’s the thyroid: a fundamental and clinical text. 8th ed. Philadelphia: Lippincott Williams & Wilkins; 2000. p. 7–19.

    Google Scholar 

  25. Clifton-Bligh RJ, Wentworth JM, Heinz P, Crisp MS, John R, Lazarus JH, 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 

  26. Prakash R, Narayanan RV, Chakravarty SK. Differentiation between suppressed thyroid tissue and thyroid hemiagenesis with Tc-99 m pertechnetate radio-nuclide angiography. Clin Nucl Med. 1990;15:605–9.

    Article  CAS  PubMed  Google Scholar 

  27. Veliz J, Pineda G. Thyroid hemiagenesis associated with Basedow-Graves disease: report of a case. Rev Med Chil. 2000;128:896–8.

    CAS  PubMed  Google Scholar 

  28. Baldini M, Orsatt IA, Cantalamessa L. A singular case of Graves’ disease in congenital thyroid hemiagenesis. Horm Res. 2005;63:107–10.

    Article  CAS  PubMed  Google Scholar 

  29. Sasaki H, Futata T, Ninomiya H, Higashi Y, Okumura M. CT and MR imagings of single thyroid lobe (thyroid hemiagenesis) with Graves’ disease. Postgrad Med J. 1991;67:701.

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  30. Monroy-Santoyo S, Ibarra-González I, Fernández-Lainez C, Greenawalt-Rodríguez S, Chacón-Rey J, Calzada-León R, et al. Higher incidence of thyroid agenesis in Mexican newborns with congenital hypothyroidism associated with birth defects. Early Hum Dev. 2012;88:61–4.

    Article  PubMed  Google Scholar 

  31. Vaswani K, Vitellas KM, Bennett WF. Thyroid hemiagenesis with adenoma. AJR Am J Roentgenol. 2000;175:895–8.

    Article  CAS  PubMed  Google Scholar 

  32. Sharma R, Mondal A, Popli M, Sahoo M, Malhotra N, Soni S. Hemiagenesis of the thyroid associated with chronic lymphocytic thyroiditis. Clin Nucl Med. 2001;26:506–8.

    Article  CAS  PubMed  Google Scholar 

  33. Khatri VP, Espinosa MH, Harada WA. Papillary adenocarcinoma in thyroid hemiagenesis. Head Neck. 1992;14:312–5.

    Article  CAS  PubMed  Google Scholar 

  34. Huang SM, Chen HD, Wen TY, Kun MS. Right thyroid hemiagenesis associated with papillary thyroid cancer and an ectopic prelaryngeal thyroid: a case report. J Formos Med Assoc. 2002;101:368–71.

    PubMed  Google Scholar 

  35. Pizzini AM, Papi G, Corrado S, Carani C, Roti E. Thyroid hemiagenesis and incidentally discovered papillary thyroid cancer: case report and review of the literature. J Endocrinol Invest. 2005;28:66–71.

    Article  CAS  PubMed  Google Scholar 

Download references

Conflict of interest

No conflict of interest was declared by the authors.

Author information

Authors and Affiliations

  1. Department of General Surgery, Kocaeli University School of Medicine, Izmit, Kocaeli, Turkey

    Turgay Şimşek & Nuh Zafer Cantürk

  2. Experimental Medicine and Research Laboratory, Kocaeli University School of Medicine, Izmit, Kocaeli, Turkey

    Nuh Zafer Cantürk

  3. Department of Endocrinology and Metabolism, Kocaeli University School of Medicine, Izmit, Kocaeli, Turkey

    Zeynep Cantürk

  4. Department of Pathology, Kocaeli University School of Medicine, Izmit, Kocaeli, Turkey

    Yeşim Gürbüz

  5. Experimental Research Laboratory (DETAB), Kocaeli Üniversitesi Tıp Fakültesi, Genel Cerrahi Anabilim Dalı, Umuttepe, Kocaeli, Turkey

    Turgay Şimşek & Nuh Zafer Cantürk

  6. Kocaeli Üniversitesi Tıp Fakültesi, Endokrinoloji ve Metabolizma Hastalıkları, Umuttepe, Kocaeli, Turkey

    Zeynep Cantürk

  7. Kocaeli Üniversitesi Tıp Fakültesi, Patoloji Anabilim Dalı, Umuttepe, Kocaeli, Turkey

    Yeşim Gürbüz

Authors
  1. Turgay Şimşek
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Nuh Zafer Cantürk
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Zeynep Cantürk
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Yeşim Gürbüz
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Turgay Şimşek.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Şimşek, T., Cantürk, N.Z., Cantürk, Z. et al. Bilobar thyroid agenesis with primary hyperparathyroidism: report of a case. Surg Today 45, 787–792 (2015). https://doi.org/10.1007/s00595-014-0990-5

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00595-014-0990-5

Keywords

Search

Navigation