Advertisement

Mild sporadic primary hyperparathyroidism: high rate of multiglandular disease is associated with lower surgical cure rate

  • Emmanuelle Trébouet
  • Sahar Bannani
  • Matthieu Wargny
  • Christophe Leux
  • Cécile Caillard
  • Françoise Kraeber-Bodéré
  • Karine Renaudin
  • Lucy Chaillous
  • Éric Mirallié
  • Catherine AnsquerEmail author
Original Article
  • 23 Downloads

Abstract

Background

Mild primary hyperparathyroidism (serum calcium ≤ 2.85 mmol/L) is the most representative form of pHPT nowadays. The aim of this study was to evaluate its subtypes and the multiglandular disease (MGD) rate as it may lower the sensitivity of preoperative parathyroid scintigraphy and the surgical cure rate.

Methods

We retrospectively included patients with mild pHPT who underwent parathyroid dual-tracer scintigraphy with 99mTc-MIBI SPECT/CT and surgery between January 2013 and December 2015. Cure was defined as normalization of serum calcium (or PTH in the normocalcemic form) at 6 months. MGD was defined by either two abnormal resected glands or persistent disease after resection of at least one abnormal gland.

Results

We included 121 patients. Median preoperative serum calcium was 2.68 mmol/L and median PTH was 83.4 pg/mL. A total of 141 glands were resected (95 adenomas, 33 hyperplasias). The subtypes were 57% classic, 32.2% normohormonal, and 10.7% normocalcemic. MGD occurred in 23.5% of patients divided as 13%, 30%, and 64% respectively (p = 0.0011). The surgical cure rate was 85.2%.

The normocalcemic form had lower cure rate than the normohormonal (45% vs 84%, p = 0.018) and classic forms (45% vs 93%, p = 0.0006). MIBI scintigraphy identified at least one abnormal lesion, later confirmed by the pathologist in 90/98 patients, making the sensitivity per patient 91.8% (95% CI 84.1–96.2%).

Conclusions

MGD is strongly associated with mild pHPT, especially the normocalcemic form where it accounts for 64% of cases. Bilateral neck exploration should be performed in this population to improve the cure rate, even if the scintigraphy shows a single focus.

Keywords

Mild primary hyperparathyroidism Multiglandular disease Parathyroidectomy MIBI scintigraphy Neck ultrasound Normocalcemic form Sestamibi scan 

Notes

Authors’ contributions

Dr. Emmanuelle Trébouet contributed to the study conception and design, acquisition of the data, analysis and interpretation of the data, drafting of the manuscript, and critical revision of the manuscript.

Dr. Sahar Bannani contributed to the study conception and design, analysis and interpretation of the data, drafting of manuscript, and critical revision of the manuscript.

Dr. Matthieu Wargny contributed to the acquisition of the data, analysis and interpretation of the data, drafting of the manuscript, and critical revision of the manuscript.

Dr. Christophe Leux contributed to the study conception and design, acquisition of the data, analysis and interpretation of the data, and critical revision of the manuscript.

Dr. Cécile Caillard contributed to the study conception and design, acquisition of the data, analysis and interpretation of the data, drafting of the manuscript, and critical revision of the manuscript.

Prof. Françoise Kraeber-Bodéré contributed to the study conception and design, acquisition of the data, analysis and interpretation of the data, drafting of the manuscript, and critical revision of the manuscript.

Dr. Karine Autain-Renaudin contributed to the acquisition of the data, analysis and interpretation of the data, and critical revision of manuscript.

Dr. Lucy Chaillous contributed to the study conception and design, acquisition of the data, analysis and interpretation of the data, and critical revision of the manuscript.

Prof. Eric Mirallié contributed to the study conception and design, acquisition of the data, analysis and interpretation of the data, drafting of the manuscript, and critical revision of the manuscript.

Dr. Catherine Ansquer contributed to the study conception and design, acquisition of the data, analysis and interpretation of the data, drafting of the manuscript, and critical revision of the manuscript.

Compliance with ethical standards

Conflict of interest

All authors declare that they have no conflict of interest.

Informed consent

Informed consent was obtained from all individual participants included in the study.

Ethical approval

All procedures performed were in accordance with the ethical standards of the institutional, the national research committee and with the 1964 Helsinki Declaration and its later amendments.

Supplementary material

423_2019_1782_MOESM1_ESM.docx (40 kb)
ESM 1 (DOCX 39 kb)

References

  1. 1.
    Bilezikian JP, Khan AA, Potts JT (2009) Guidelines for the Management of Asymptomatic Primary Hyperparathyroidism: Summary Statement from the Third International Workshop. J Clin Endocrinol Metab 94:335–339CrossRefGoogle Scholar
  2. 2.
    Barczyński M, Bränström R, Dionigi G, Mihai R (2015) Sporadic multiple parathyroid gland disease—a consensus report of the European Society of Endocrine Surgeons (ESES). Langenbeck's Arch Surg 400:887–905CrossRefGoogle Scholar
  3. 3.
    Low RA, Katz AD (1998) Parathyroidectomy via bilateral cervical exploration: a retrospective review of 866 cases. Head Neck 20:583–587CrossRefGoogle Scholar
  4. 4.
    Bilezikian JP, Brandi ML, Eastell R, Silverberg SJ, Udelsman R, Marcocci C, Potts JT Jr (2014) Guidelines for the management of asymptomatic primary hyperparathyroidism: summary statement from the Fourth International Workshop. J Clin Endocrinol Metab 99:3561–3569CrossRefGoogle Scholar
  5. 5.
    Silverberg SJ, Clarke BL, Peacock M, Bandeira F, Boutroy S, Cusano NE, Dempster D, Lewiecki EM, Liu JM, Minisola S, Rejnmark L, Silva BC, Walker MD, Bilezikian JP (2014) Current issues in the presentation of asymptomatic primary hyperparathyroidism: proceedings of the Fourth International Workshop. J Clin Endocrinol Metab 99:3580–3594CrossRefGoogle Scholar
  6. 6.
    Caillard C, Sebag F, Mathonnet M, Gibelin H, Brunaud L, Loudot C, Kraimps JL, Hamy A, Bresler L, Charbonnel B, Leborgne J, Henry JF, Nguyen JM, Mirallié E (2007) Prospective evaluation of quality of life (SF-36v2) and nonspecific symptoms before and after cure of primary hyperparathyroidism (1-year follow-up). Surgery 141:153–159; discussion 159-160 CrossRefGoogle Scholar
  7. 7.
    Blanchard C, Mathonnet M, Sebag F, Caillard C, Kubis C, Drui D, van Nuvel E, Ansquer C, Henry JF, Masson D, Kraeber-Bodéré F, Hardouin JB, Zarnegar R, Hamy A, Mirallié E (2014) Quality of life is modestly improved in older patients with mild primary hyperparathyroidism postoperatively: results of a prospective multicenter study. Ann Surg Oncol 21:3534–3540CrossRefGoogle Scholar
  8. 8.
    Blanchard C, Mathonnet M, Sebag F, Caillard C, Hamy A, Volteau C, Heymann MF, Wyart V, Drui D, Roy M, Cariou B, Archambeaud F, Rodien P, Henry JF, Zarnegar R, Hardouin JB, Mirallié E (2013) Surgery for ‘asymptomatic’ mild primary hyperparathyroidism improves some clinical symptoms postoperatively. Eur J Endocrinol 169:665–672CrossRefGoogle Scholar
  9. 9.
    Lim JY, Herman MC, Bubis L, Epelboym I, Allendorf JD, Chabot JA, Lee JA, Kuo JH (2017) Differences in single gland and multigland disease are seen in low biochemical profile primary hyperparathyroidism. Surgery 161:70–77CrossRefGoogle Scholar
  10. 10.
    Paek SH, Kim S-J, Choi JY, Lee KE (2018) Clinical usefulness of intraoperative parathyroid hormone monitoring for primary hyperparathyroidism. Ann Surg Treat Res 94:69–73CrossRefGoogle Scholar
  11. 11.
    Cayo AK, Sippel RS, Schaefer S, Chen H (2009) Utility of intraoperative PTH for primary hyperparathyroidism due to multigland disease. Ann Surg Oncol 16:3450–3454CrossRefGoogle Scholar
  12. 12.
    Applewhite MK, Schneider DF (2014) Mild primary hyperparathyroidism: a literature review. Oncologist 19:919–929CrossRefGoogle Scholar
  13. 13.
    Wong KK, Fig LM, Gross MD, Dwamena BA (2015) Parathyroid adenoma localization with 99mTc-sestamibi SPECT/CT: a meta-analysis. Nucl Med Commun 36:363–375CrossRefGoogle Scholar
  14. 14.
    Ansquer C, Mirallié E, Carlier T et al (2008) Preoperative localization of parathyroid lesions. Value of 99mTc-MIBI tomography and factors influencing detection. Nukl Nucl Med 47:158–162CrossRefGoogle Scholar
  15. 15.
    Medas F, Erdas E, Longheu A, Gordini L, Pisano G, Nicolosi A, Calò PG (2016) Retrospective evaluation of the pre- and postoperative factors influencing the sensitivity of localization studies in primary hyperparathyroidism. Int J Surg Lond Engl 25:82–87CrossRefGoogle Scholar
  16. 16.
    Swanson TW, Chan SK, Jones SJ, Bugis S, Irvine R, Belzberg A, Levine D, Wiseman SM (2010) Determinants of Tc-99m sestamibi SPECT scan sensitivity in primary hyperparathyroidism. Am J Surg 199:614–620CrossRefGoogle Scholar
  17. 17.
    Carlier T, Oudoux A, Mirallié E, Seret A, Daumy I, Leux C, Bodet-Milin C, Kraeber-Bodéré F, Ansquer C (2008) 99mTc-MIBI pinhole SPECT in primary hyperparathyroidism: comparison with conventional SPECT, planar scintigraphy and ultrasonography. Eur J Nucl Med Mol Imaging 35:637–643CrossRefGoogle Scholar
  18. 18.
    Bilezikian JP (2018) Primary hyperparathyroidism. J Clin Endocrinol Metab 103:3993–4004CrossRefGoogle Scholar
  19. 19.
    Rosen CJ, Gallagher JC (2011) The 2011 IOM report on vitamin D and calcium requirements for North America: clinical implications for providers treating patients with low bone mineral density. J Clin Densitom Off J Int Soc Clin Densitom 14:79–84CrossRefGoogle Scholar
  20. 20.
    Michaud L, Burgess A, Huchet V, Lefèvre M, Tassart M, Ohnona J, Kerrou K, Balogova S, Talbot JN, Périé S (2014) Is 18F-fluorocholine-positron emission tomography/computerized tomography a new imaging tool for detecting hyperfunctioning parathyroid glands in primary or secondary hyperparathyroidism? J Clin Endocrinol Metab 99:4531–4536CrossRefGoogle Scholar
  21. 21.
    Kluijfhout WP, Vorselaars WMCM, van den Berk SAM, Vriens MR, Borel Rinkes IHM, Valk GD, van Dalen T, de Klerk JMH, de Keizer B (2016) Fluorine-18 fluorocholine PET-CT localizes hyperparathyroidism in patients with inconclusive conventional imaging: a multicenter study from the Netherlands. Nucl Med Commun 37:1246–1252CrossRefGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2019

Authors and Affiliations

  • Emmanuelle Trébouet
    • 1
  • Sahar Bannani
    • 2
  • Matthieu Wargny
    • 1
  • Christophe Leux
    • 3
  • Cécile Caillard
    • 2
  • Françoise Kraeber-Bodéré
    • 4
    • 5
  • Karine Renaudin
    • 6
  • Lucy Chaillous
    • 1
  • Éric Mirallié
    • 2
  • Catherine Ansquer
    • 4
    • 5
    Email author
  1. 1.Service d’Endocrinologie, CHU de NantesNantes Cedex 1France
  2. 2.Clinique de Chirurgie Digestive et Endocrinienne, CHU de NantesHôtel DieuNantes Cedex 1France
  3. 3.Service d’Information Médicale, CHU de NantesNantes Cedex 1France
  4. 4.Service de Médecine Nucléaire, CHU de NantesHôtel DieuParisFrance
  5. 5.CRCINA, INSERM, CNRSUniversité d’Angers, Université de NantesNantes Cedex 1France
  6. 6.Service d’Anatomie Cytologie Pathologique, CHU de NantesHôtel DieuNantes Cedex 1France

Personalised recommendations