Leydig cell tumor of the testis with azoospermia and elevated delta4 androstenedione: case report.
BACKGROUND
Secreting interstitial cell (Leydig cell) tumors are rare. In adults, the clinical picture and steroid levels are variable.


METHODS
This paper presents a case of left testicular tumor, showing azoospermia with normal serum level of total testosterone, collapsed FSH and LH, and high delta4 androstenedione. Histopathological investigation revealed a Leydig cell tumor. TESE allowed spermatozoa extraction and freezing. Testicular histology found hypospermatogenesis and germ-cell aplasia with interstitial fibrosis. Surgical resection of the tumor resulted in normalization of gonadotropins and fall in serum delta4 androstenedione to subnormal levels in the postoperative period confirming that the tumor was secreting delta4 androstenedione. It was hypothesized that high delta4 androstenedione resulted in intra tumoral 17 β-HSD overtaken by delta4 androstenedione or that 17 β-HSD activity in the tumor was different from that of normal Leydig cells. Three months after surgery sperm analysis found a complete recovery of spermatogenesis. A spontaneous pregnancy occurred 3 months after surgery and a girl was born.


CONCLUSIONS
In this case, the diagnosis of testicular Leydig cell tumor secreting delta4 androstenedione was made in a context of azoospermia.
Department of Andrology, Lille University Hospital, Lille, France ; Department of Andrology, CHRU Lille, Hôpital Calmette, Boulevard du Professeur Leclercq, 59037 Lille Cedex, France.
J
J
Prasivoravong
Biology of Reproduction Unit, Lille University Hospital, Lille, France ; EA4308 Gametogenesis and Gamete Quality, University of Lille, Lille, France.
A-L
AL
Barbotin
Department of Andrology, Lille University Hospital, Lille, France.
A
A
Derveaux
Department of Andrology, Lille University Hospital, Lille, France.
C
C
Leroy
Department of Pathology, Lille University Hospital, Lille, France.
X
X
Leroy
Department of Radiology, Lille University Hospital, Lille, France.
P
P
Puech
Biology of Reproduction Unit, Lille University Hospital, Lille, France ; EA4308 Gametogenesis and Gamete Quality, University of Lille, Lille, France.
V
V
Mitchell
Department of Andrology, Lille University Hospital, Lille, France ; EA4308 Gametogenesis and Gamete Quality, University of Lille, Lille, France.
F
F
Marcelli
Department of Andrology, Lille University Hospital, Lille, France ; EA4308 Gametogenesis and Gamete Quality, University of Lille, Lille, France.
J-M
JM
Rigot
Azoospermia; Hormone secreting testicular tumor; Infertility; Leydig cell tumor; TESE; delta4 androstenedione