Recurrent Severe Hypoglycemia Secondary to Benign Phyllodes Tumour of the Breast : A Rare Case of Non-Islet Cell Tumor-induced Hypoglycemia (NICTH)

Hwee Ching Tee; Vijiya Mala  Valayatham

doi:10.15605/jafes.036.02.16

Authors

Hwee Ching Tee Putrajaya Hospital, Malaysia
Vijiya Mala Valayatham Putrajaya Hospital, Malaysia https://orcid.org/0000-0001-6750-6557

DOI:

https://doi.org/10.15605/jafes.036.02.16

Keywords:

hypoglycemia, insulin-like Growth Factor II, phyllodes tumor, mastectomy, corticosteroids

Abstract

Non-Islet Cell Tumor-induced hypoglycemia (NICTH) secondary to phyllodes tumour is extremely rare but potentially life threatening if not treated promptly. We report a case of a 46-year-old female without underlying diabetes mellitus who presented with a large breast tumor and recurrent severe symptomatic hypoglycemia. Investigations supported the diagnosis of NICTH and the hypoglycemia only resolved after corticosteroids and mastectomy. This case highlights the importance of considering NICTH in the evaluation of patients with voluminous tumour and hypoglycemia.

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Author Biographies

Hwee Ching Tee, Putrajaya Hospital, Malaysia

Endocrine Unit

Vijiya Mala Valayatham, Putrajaya Hospital, Malaysia

Endocrine Unit

References

Ben Hassouna J, Damak T, Gamoudi A, et al. Phyllodes tumors of the breast: A case series of 106 patients. Am J Surg. 2006;192(2):141-7. https://pubmed.ncbi.nlm.nih.gov/16860620. https://doi.org/10.1016/j.amjsurg.2006.04.007.

Reinfuss M, Mituś J, Duda K, Stelmach A, Ryś J, Smolak K. The treatment and prognosis of patients with phyllodes tumor of the breast: An analysis of 170 cases. Cancer. 1996;77(5):910-6. https://pubmed.ncbi.nlm.nih.gov/8608483. https://doi.org/10.1002/(sici)1097-0142(19960301)77:5<910::aid-cncr16>3.0.co;2-6.

Dynkevich Y, Rother KI, Whitford I, et al. Tumors, IGF-2, and hypoglycemia: Insights from the clinic, the laboratory, and the historical archive. Endocr Rev. 2013;34(6):798-826. https://pubmed.ncbi.nlm.nih.gov/23671155. https://doi.org/10.1210/er.2012-1033.

Li TC, Reed CE, Stubenbord WT Jr, et al. Surgical cure of hypoglycemia associated with cystosarcoma phyllodes and elevated nonsuppressible insulin-like protein. Am J Med. 1983;74(6):1080-4. https://pubmed.ncbi.nlm.nih.gov/6305192. https://doi.org/10.1016/0002-9343(83)90823-9.

Tanaka S, Muramoto Y,Sakae K, Sato E. Giant malignant phyllodes tumor of the breast with hypoglycemia and left hemiplegia; Report of a case. Nippon Byori Gakkai Kaishi 1986; 75:481-2 (abstr) (in Japanese).

Bleau B, Vlahos W, Guyga H. High molecular weight insulin-like growth factor-II (HMW IGF-II) associated with fasting hypoglycemia and a phyllodes tumor of the breast. Clin Invest Med 1991;14(Suppl.):36 (abstr).

Ishido H, Kitaoka M, Uchigata M, Kaji M, Maeda M, Nagayama T. A case of phyllodes tumor ofthe breast producing IGF-II. Nippon Naibunpitsu Gakkai Zashi 1992;68:825 (abstr) (in Japanese).

Miura K, Noma M, Ishikawa K, Katayama S. Giant breast tumor (cystosarcoma phyllodes) associated with unconsciousness by persistent hypoglycemia; Report of an autopsied case. Iryou 1992; 46:179 (abstr) (in Japanese).

Kataoka T, Haruta R, Goto T, et al. Malignant phyllodes tumor of the breast with hypoglycemia: report of a case. Jpn J Clin Oncol. 1998;28(4):276-80. https://pubmed.ncbi.nlm.nih.gov/9657015. https://doi.org/ 10.1093/jjco/28.4.276.

Aguiar Bujanda D, Rivero Vera JC, Cabrera Suárez MA, et al. Hypoglycemic coma secondary to big insulin-like growth factor II secretion by a giant phyllodes tumor of the breast. Breast J. 2007;13(2):189-91. https://pubmed.ncbi.nlm.nih.gov/17319862. https://doi.org/10.1111/j.1524-4741.2007.00398.x.

Hino N, Nakagawa Y, Ikushima Y, Yoshida M, Tsuyuguchi M. A case of a giant phyllodes tumor of the breast with hypoglycemia caused by high-molecular-weight insulin-like growth factor II. Breast Cancer. 2010;17(2):142-5. https://pubmed.ncbi.nlm.nih.gov/19259766. https://doi.org/10.1007/s12282-009-0094-z.

Renard E, Langbour-Remy C, Klein M, Le Bouc Y, Weryha G, Cuny T. Severe hypoglycemia with "Big"-IGF-2 oversecretion by a giant phyllode tumor of the breast: a rare case of non-islet cell tumor-induced hypoglycemia (NICTH). Ann Endocrinol (Paris). 2012;73(5):488-91. https://pubmed.ncbi.nlm.nih.gov/22867750. https://doi.org/10.1016/j.ando.2012.04.011.

Agrawal S, Nair R, Malali B, Nayal B. Case of benign phyllodes tumour associated with hypoglycemia. Malays J Med Sci. 2013;20(2):67-9. https://pubmed.ncbi.nlm.nih.gov/23983580. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3744002.

Pacioles T, Seth R, Orellana C, John I, Panuganty V, Dhaliwal R. Malignant phyllodes tumor of the breast presenting with hypoglycemia: A case report and literature review. Cancer Manag Res. 2014;6:467-73. https://pubmed.ncbi.nlm.nih.gov/25525388. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4266253. https://doi.org/10.2147/CMAR.S71933.

Saito Y, Suzuki Y, Inomoto C, et al. A case of giant borderline phyllodes tumor of the breast associated with hypoglycemia. Tokai J Exp Clin Med. 2016;41(3):118-22. https://pubmed.ncbi.nlm.nih.gov/27628602.

Hikichi M, Kiriyama Y, Hayashi T, et al. A hypoglycemia-inducing giant borderline phyllodes tumor secreting high-molecular-weight insulin-like growth factor II: Immunohistochemistry and a Western Blot Analysis. Intern Med. 2018;57(2):237-41. https://pubmed.ncbi.nlm.nih.gov/29021486. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5820043. https://doi.org/10.2169/internalmedicine.9287-17.

Zhao J, Gao M, Ren Y, Cao S, Wang H, Ge R. A giant borderline phyllodes tumor of breast with skin ulceration leading to non-insular tumorigenic hypoglycemia: A case report and literature review. Front Endocrinol (Lausanne). 2021;12:651568. https://pubmed.ncbi.nlm.nih.gov/33841338. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8027481. https://doi.org/10.3389/fendo.2021.651568.

Guillot E, Couturaud B, Reyal F, et al. Management of phyllodes breast tumors. Breast J. 2011;17(2):129-37. https://pubmed.ncbi.nlm.nih.gov/21251125. https://doi.org/10.1111/j.1524-4741.2010.01045.x.

Fukuda I, Hizuka N, Ishikawa Y, Yasumoto K, et al. Clinical features of insulin-like growth factor-II producing non-islet-cell tumor hypoglycemia. Growth Horm IGF Res. 2006;16(4):211-6. https://pubmed.ncbi.nlm.nih.gov/16860583. https://doi.org/10.1016/j.ghir.2006.05.003.

Bodnar TW, Acevedo MJ, Pietropaolo M. Management of non-islet-cell tumor hypoglycemia: A clinical review. J Clin Endocrinol Metab. 2014;99(3):713-22. https://pubmed.ncbi.nlm.nih.gov/24423303. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5393479. https://doi.org/10.1210/jc.2013-3382.

Hizuka N, Fukuda I, Takano K, Okubo Y, Asakawa-Yasumoto K, Demura H. Serum insulin-like growth factor II in 44 patients with non-islet cell tumor hypoglycemia. Endocr J. 1998;45 Suppl:S61-5. https://pubmed.ncbi.nlm.nih.gov/9790231. https://doi.org/10.1507/endocrj.45.suppl_s61.

Cryer PE, Axelrod L, Grossman AB, et al. Evaluation and management of adult hypoglycemic disorders: An Endocrine Society Clinical Practice Guideline. J Clin Endocrinol Metab. 2009;94(3):709-28. https://pubmed.ncbi.nlm.nih.gov/19088155. https://doi.org/10.1210/jc.2008-1410.