Addison's disease presenting as male infertility.
OBJECTIVE
To present a case of primary male infertility with severe oligospermia and hyperpigmented skin as the main presenting clinical signs.


METHODS
Case report.


METHODS
University-affiliated teaching hospital.


METHODS
A 32-year-old male with severe oligospermia and his 31-year-old female partner with normal cycles.


METHODS
Hydrocortisone, fludrocortisone, methimazole, and eventual IVF.


METHODS
Improved semen analysis, correction of Addison's disease, correction of hyperthyroidism, and ART pregnancy.


RESULTS
Severe oligospermia of 5 million per milliliter with 15% motility and ACTH level of 2,800 pg/mL on presentation. Endocrinology evaluation revealed cortisol of <0.1 microg/dL, and Cortrosyn test showed no response (Addison's disease). The patient was incidentally found to have a thyroid stimulating hormone level of <0.1 microIU/mL, T4 of 9.5 microg/dL, Free T4 Index calculation of 11.6 microg/dL, and quantitative triiodothyronine of 273 ng/dL on presentation. His Addison's disease was managed with hydrocortisone and fludrocortisone, and his hyperthyroidism, with methimazole. Semen parameters increased to 34 million per milliliter with 45% motility and 5% Kruger morphology 1 year after initial presentation. Hormone parameters normalized. Rather than allowing for more time for natural conception or IUI, the couple decided to proceed with IVF because of insurance coverage before semen parameters normalized.


CONCLUSIONS
This is the first reported case of Addison's disease presenting as male infertility with hyperpigmentation of the skin being the only other presenting sign. The underlying etiology of the Addison's in this case remains uncertain, but it is presumed because of autoimmunity. Addison's disease may be associated with hyperthyroidism, and hyperthyroidism may have contributed to the oligospermia. This case highlights why men with abnormal semen parameters should be thoroughly evaluated before proceeding with assisted reproduction.
Dartmouth Medical School, Hanover, New Hampshire, USA. bfk@hitchcock.org
Brian F
BF
Kowal
John
J
Turco
Ajay K
AK
Nangia
D000224:Addison Disease / Q000175:diagnosis* / Q000188:drug therapy; D000328:Adult; D003937:Diagnosis, Differential; D006801:Humans; D007248:Infertility, Male / Q000175:diagnosis* / Q000188:drug therapy; D008297:Male; D008713:Methimazole / Q000627:therapeutic use
D008713:Methimazole