Case Study Reports Successful Strategy for Infertility Caused by Testicular Sarcoidosis

Testicular sperm aspiration for in vitro fertilization (IVF) is a successful strategy to overcome infertility associated with methotrexate– and steroid-resistant testicular sarcoidosis, a case study shows.

The report documents a case of a man who, within two years of a sarcoidosis diagnosis, showed complete azoospermia (absence of sperm in semen), likely associated with a testicular granuloma blocking sperm passage. This case highlighted that obstructive azoospermia should be considered in men diagnosed with sarcoidosis who want to have children, the researchers noted.

The study, “Spontaneous Infertility Secondary to Testicular Sarcoidosis: A Case Report,” was published in the Cureus Journal of Medical Medicine.

Sarcoidosis-related chronic inflammation leads to the formation of small abnormal lumps or nodules — called granulomas — in several organs of the body, most often in the lungs. Involvement of genital and urinary organs (genitourinary sarcoidosis) is very rare.

Previous studies have shown that the testes were affected in about half of the patients with reproductive tract involvement and that testicular sarcoidosis can lead to infertility. While many reported cases of sarcoidosis-associated infertility were resolved with some form of steroid treatment, others were not.

Now, researchers at the Miami-based IVFMD fertility center and Lake Erie College of Osteopathic Medicine’s Research reported the case of a man with infertility associated with methotrexate- and steroid-resistant testicular sarcoidosis.

The man and his partner went to the fertility clinic two years after successfully conceiving their first child naturally, with subsequent infertility. Since then, he had been diagnosed with sarcoidosis and started treatment with Solu-Medrol (methylprednisolone injection, an anti-inflammatory steroid), methotrexate, and levaquin (an antibiotic).

Two separate semen analyses six weeks apart showed azoospermia. Since the couple had conceived four times before infertility was diagnosed (three of the pregnancies ended in spontaneous abortions), genetic factors were ruled out as a potential cause of azoospermia.

Considering potential methotrexate-induced male infertility, the man was counseled to discontinue methotrexate treatment. However, three months after he stopped treatment, a semen analysis revealed no improvement.

Lab work showed that his hormone levels, including testosterone, thyroid stimulating hormone, prolactin, follicle stimulating hormone, and luteinizing hormone were all within normal limits.

Overall, the evidence suggested obstructive azoospermia, meaning that sperm was likely being normally produced but something was blocking its passage.

The man was then referred to a urologist to be considered for testicular sperm aspiration — the collection of sperm cells from the testicle through a small needle — for IVF. On examination, the urologist reported a palpable testicular nodule, identifying it as a granuloma.

However, “due to loss of follow-up, a confirmatory biopsy of the testicular nodule was never reported by the urology team,” the researchers wrote.

Sperm aspiration followed by IVF was found to be an effective approach to manage the man’s infertility, as it resulted in the conception and delivery of a healthy boy at term.

“This case report offers an insight into successful fertility treatment of methotrexate and steroid-resistant testicular sarcoidosis,” the researchers wrote, adding that “the possibility of obstructive azoospermia should be considered in males diagnosed with sarcoidosis who are seeking to preserve their reproductive potential.”

The team noted that many cases of genitourinary sarcoidosis reported to date show subtle, if any, symptoms which may challenge its diagnosis especially if patients are not attempting to have children naturally.

“Screening male patients with an initial semen analysis upon diagnosis of sarcoidosis would be a low-cost and effective measure to both screen and evaluate for future genitourinary involvement,” the researchers wrote.

Further studies are needed to assess the real frequency of genitourinary involvement in sarcoidosis patients, the team noted.