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Androblastoma of the Testicle: Report of a Case in an Infant 3½ Months Old
THE JoGRYAL OF lJRULOGY
V0!. 7\J, No. 5, May 1958 Ftinled 'in C.S.A.
AKDROBLASTOl\IA OF THE TESTICLE: REPORT OF A CASE lN A~ INFANT :'3}:i MONTHS OLD AZIZ FAM
AND
KAMAL G. ISHAK*
From the Deparlmenl of L'rolouy, Faculty of Medicine, Cairo University, E(Jypt and Department <~l Palholo(Jy, U.S. Naval illedical Research Unit No. B, Cairo, Eyypt
Patie11t R.ivl.S., an Egyptian male infant three and one half months old, wa,; reported by his mother to shmv a progressive increase in the :c;ize of hi;; left testicle of one month's duration. Pain had not been present, as evidenced by the absence of any unusual crying. A general practitioner had already :,:een the child and had prescribed a fortnight's course of penicillin and ::,treptomycin without any apparent diminution in the size of the ::,welling. OH examination it was noted that the general condition of the child was exeelleut. The pube, temperature, :,;kin and mucou:,; membrane:,; ,rnrc within normal limits. The breasb were not enlarged. There was no lymphadenopathy in the axillae, supraelavicular regions or groin:,;. The lungs and heart wenc normal to palpation, percussion and auscultation. Abdominal palpation revealed no ascites or intra-abdominal :c;welling. Local exan1ination of the Hcrotum showed enlargement of the left :,;ide, but no impulse was present when the child cried. The :,;welling was wholly intrascrotal, was m al in shape, and was about 5 cm. in it:o greatest dimension. The surface was smooth with a few indistinct bosses, and the consic1tency firm. A small amount of fluid was detected by pinching. The epididymi,; could not be palpated separately from the s,velling. Thickening of the cord was noted, but there was no matting of the cord structures and no ma:,;ses could be felt. The intrascrotal content::; on the right :oide were within normal limits. The Rize of the penis corresponded to the age of the patient. Laboratory analyse:,;, including a VI/ a:,;::,ermann reaction, revealed negative finding:,;. Hormonal assays ,rnre not attempted in view of the fact that :,;igns of endocrinal imbalance were not present. A tumor of the testicle wa:-; suspected and consequently a left orchiectomy was performed, ,vith excision of the cord as far as the internal abdominal ring. The ,rnund healed finst intention in seven days and the child ,vas discharged fron, the ho:opital. He ha:,; been regularly followed up for a period of hrn years and i,; alive and in good health up to the present time. The gros:,; specimen (fig. 1) consisted of the disea:,;ed testicle, the epididymis and the spermatic: cord. The enlarged tec1tiele mea:,;ured 3}~ by 2 by 2 cm. and was inveHted -with au intact tunica vaginalis. A small amount of clear, strawcolored fluid fimrnd out when the parietal layer was incic1ed. The viseeral surface of the tunica vaginalis ,vas smooth and gli:,;tening. Section of the te:,;ticle rnvealed that itH consistency ,ya:,; firm. The cut :,;urface -was opaque, homogeneom,, and greyish ,vhite in color. The normal structure of the testicle wati not presento Distinct lobulation was noted, the lobules being separated by depressed fibrou::; trabeculae, and memmring between 0.5 and 0.7 cm. in diameter. No areas of 0
Accepted for publication June 1:3, HJ57. * Present address: Bapti~t Memorial Hospital, San Antonio, Texas.
859
860
AZIZ FAM AND KAMAL G
ISHAK
necrosis or hemorrhage were observed. The cord, though somewhat thickened, and the epididymis were within normal limits. Microscopically, the sections showed a somewhat thickened tunica vaginalis. Immediately subjacent to the tunica albuginea some compressed tubules, otherwise without note, were observed (fig. 2, A). Occupying the place of the normal testicle was a well-encapsulated tumor, partitioned into lobules by fibrous connective tissue septa, and infiltrated with some lymphocytes, polymorphs and
Fm. 1. Gross specimen removed by operation
Fm. 2. A, tumor seen to be well encapsulated. Few compressed, normal tubules are noted between collagen bundles (low magnification). B, predominantly stromal element is present here, though occasional attempts at formation of lumina can be seen.
ANDROBLASTOMA OF TESTICLI~
8G1
Fm. 3 . .4, predominantly tubular element is observed in this section of tumor. B, higher magnification to show structure of tubules which are well demarcated by base· ment membrane,
eosinophil cells. The stroma of the lobules was densely packed with round and polygonal cells, with a scant, lightly staining cytoplasm (fig. 2, B). The nuclei were round or oval, showed a fine granular chromatin network, and one or two small nucleoli. Mitotic figures were observed occasionally throughout. A defi .. nitely tubular pattern was present in some areas (fig. :')). The tubules had a definite basement membrane, and were lined by one or more layers of euboidal epithelial cells. Shedding of epithelial cells into the lumen of some of the tubules was noted. A pale-staining eosinophilic, amorphous material was occasionally present in the larger tubules. While the stromal and tubular elements were distinct, gradations between the two were also seen, with arrangements of the cells into cords and pseudo-tubules. Leydig-like cells with vacuoles or a foamy cytoplasm were also present. Frozen sections stained with Sudan '.:\ showed the presence of fat in the Leydig cells. DISCUSSION
The androblastoma was first introduced by Teilum 1 for tumors of the testicle homologous to arrhenoblastomas (Meyer) 2 of the ovary. According to Friedman and l\1-oore, 3 they constitute 0.4 per cent of testicular tumors. are tumors originating from a testicular blastoma in which a differentiation 1 Teilum, G.: Arrhenoblastoma~androblastoma; homologous ovarian and testicular tumors; including the HO-called "luteomas" and "adrenal tumors" of ovary and interstitial cell tumors of testis. Acta Path. et JVIicrobiol. Scandinav., 23: 252--64, 1946. 2 Meyer, R.: Tubulare (testiculare) und solide -Formen der Andreiblastoma ovarii und ihre Beziehung zur Vermiinnlichung. Beitr. z. Path. Anat. u.z. a.Ilg. Path., 84: 48,5--520, 1930. "Friedman, N. B. and :VIoore, R. A.: Tumors of t,he testis. A report of fl22 cases. Military Surg., 99: 573-93, lfl46.
862
AZIZ FAM A:"
ISHAK
tendency in the direction of Sertoli and/ or Leydig cells may take place.1· 4 Accordingly, these tumors are classified histologically into: 1) tubular (tubular adenoma of Pick) 5 ; 2) mixed, and 3) diffuse stromal types. The reader is referred to the excellent account of the histopathology of these tumors by Dixon and Moore. 6 To our knowledge only six diffuse and mixed androblastomas, three of Teilum,1 · 4 and three of Freidman and JVIoore,3 have been reported, to which cases we are adding a seventh. The age incidence of the published cases varied from 4 to 52 years. Our case of an infant aged 3½ months is the youngest so far reported. The arrhenoblastomas of the ovary produce virilizing effects in females, while the homologous androblastomas in the male are sometimes associated with feminizing effects. According to Teilum,1· 4 · 7 this apparently paradoxical behavior depends on the predominance of either lipoidal androgen-producing Leydig cells (stromal element), or of estrogen-producing Sertoli cells (tubular epithelial element). Of the six reported cases referred to above, only one (Teilum's) was associated with hormonal effects, viz., gynecomastia. Our patient did not show any signs of hormonal disturbance, though admittedly, hormonal assays were not performed. None of the reported cases was malignant, although as Dixon and Moore 6 point out, the possibility exists of malignant variants of androblastoma, as indicated by the not infrequent occurrence of malignant arrhenoblastomas. SUMMARY
The history and clinical picture of a case of androblastoma of the left testis in an infant 33'1 months old are described. The tumor presented as a painless swelling of the left side of the scrotum of 1 month's duration prior to admission. Signs of hormonal imbalance were absent. A left orchiectomy was performed, the child remaining alive and well until the present time. The gross and microscopic findings are described. The tumor was of the mixed type of androblastoma, with the presence of both tubular and diffuse stromal elements. A brief comment is made on the reported cases of androblastoma in the literature. 4 Teilum, G.: Estrogen-producing Sertoli cell tumors (androblastoma tubulare lipoides) of the human testis and ovary. Homologous ovarian and testicular tumors. III. J. Clin. Endocrinol., 9: 301-18, 1949. 5 Pick, L.: Ueber Neubildungen am Genitale bei Zwittern nebst Beitragen zur Lehre von den Adenomen des Hodens und Eierstockes. Arch. f. Gyntk., 76: 191-281, 1905. 6 Dixon, F. J. and Moore, R. A.: Tumors of the male sex organs, in Atlas of Tumor Pathology, Sect. VIII~Fasc. 31b & 32, pp. 120-126, Washington D. C.: Armed Forces Institute of Pathology, 1952. 7 Teilum, G.: Endocrine varieties of "androblastoma" in ovary and testis. J. Obst. & Gynaec. Brit. EmD .. 58: 201-2, 1951.
V0!. 7\J, No. 5, May 1958 Ftinled 'in C.S.A.
AKDROBLASTOl\IA OF THE TESTICLE: REPORT OF A CASE lN A~ INFANT :'3}:i MONTHS OLD AZIZ FAM
AND
KAMAL G. ISHAK*
From the Deparlmenl of L'rolouy, Faculty of Medicine, Cairo University, E(Jypt and Department <~l Palholo(Jy, U.S. Naval illedical Research Unit No. B, Cairo, Eyypt
Patie11t R.ivl.S., an Egyptian male infant three and one half months old, wa,; reported by his mother to shmv a progressive increase in the :c;ize of hi;; left testicle of one month's duration. Pain had not been present, as evidenced by the absence of any unusual crying. A general practitioner had already :,:een the child and had prescribed a fortnight's course of penicillin and ::,treptomycin without any apparent diminution in the size of the ::,welling. OH examination it was noted that the general condition of the child was exeelleut. The pube, temperature, :,;kin and mucou:,; membrane:,; ,rnrc within normal limits. The breasb were not enlarged. There was no lymphadenopathy in the axillae, supraelavicular regions or groin:,;. The lungs and heart wenc normal to palpation, percussion and auscultation. Abdominal palpation revealed no ascites or intra-abdominal :c;welling. Local exan1ination of the Hcrotum showed enlargement of the left :,;ide, but no impulse was present when the child cried. The :,;welling was wholly intrascrotal, was m al in shape, and was about 5 cm. in it:o greatest dimension. The surface was smooth with a few indistinct bosses, and the consic1tency firm. A small amount of fluid was detected by pinching. The epididymi,; could not be palpated separately from the s,velling. Thickening of the cord was noted, but there was no matting of the cord structures and no ma:,;ses could be felt. The intrascrotal content::; on the right :oide were within normal limits. The Rize of the penis corresponded to the age of the patient. Laboratory analyse:,;, including a VI/ a:,;::,ermann reaction, revealed negative finding:,;. Hormonal assays ,rnre not attempted in view of the fact that :,;igns of endocrinal imbalance were not present. A tumor of the testicle wa:-; suspected and consequently a left orchiectomy was performed, ,vith excision of the cord as far as the internal abdominal ring. The ,rnund healed finst intention in seven days and the child ,vas discharged fron, the ho:opital. He ha:,; been regularly followed up for a period of hrn years and i,; alive and in good health up to the present time. The gros:,; specimen (fig. 1) consisted of the disea:,;ed testicle, the epididymis and the spermatic: cord. The enlarged tec1tiele mea:,;ured 3}~ by 2 by 2 cm. and was inveHted -with au intact tunica vaginalis. A small amount of clear, strawcolored fluid fimrnd out when the parietal layer was incic1ed. The viseeral surface of the tunica vaginalis ,vas smooth and gli:,;tening. Section of the te:,;ticle rnvealed that itH consistency ,ya:,; firm. The cut :,;urface -was opaque, homogeneom,, and greyish ,vhite in color. The normal structure of the testicle wati not presento Distinct lobulation was noted, the lobules being separated by depressed fibrou::; trabeculae, and memmring between 0.5 and 0.7 cm. in diameter. No areas of 0
Accepted for publication June 1:3, HJ57. * Present address: Bapti~t Memorial Hospital, San Antonio, Texas.
859
860
AZIZ FAM AND KAMAL G
ISHAK
necrosis or hemorrhage were observed. The cord, though somewhat thickened, and the epididymis were within normal limits. Microscopically, the sections showed a somewhat thickened tunica vaginalis. Immediately subjacent to the tunica albuginea some compressed tubules, otherwise without note, were observed (fig. 2, A). Occupying the place of the normal testicle was a well-encapsulated tumor, partitioned into lobules by fibrous connective tissue septa, and infiltrated with some lymphocytes, polymorphs and
Fm. 1. Gross specimen removed by operation
Fm. 2. A, tumor seen to be well encapsulated. Few compressed, normal tubules are noted between collagen bundles (low magnification). B, predominantly stromal element is present here, though occasional attempts at formation of lumina can be seen.
ANDROBLASTOMA OF TESTICLI~
8G1
Fm. 3 . .4, predominantly tubular element is observed in this section of tumor. B, higher magnification to show structure of tubules which are well demarcated by base· ment membrane,
eosinophil cells. The stroma of the lobules was densely packed with round and polygonal cells, with a scant, lightly staining cytoplasm (fig. 2, B). The nuclei were round or oval, showed a fine granular chromatin network, and one or two small nucleoli. Mitotic figures were observed occasionally throughout. A defi .. nitely tubular pattern was present in some areas (fig. :')). The tubules had a definite basement membrane, and were lined by one or more layers of euboidal epithelial cells. Shedding of epithelial cells into the lumen of some of the tubules was noted. A pale-staining eosinophilic, amorphous material was occasionally present in the larger tubules. While the stromal and tubular elements were distinct, gradations between the two were also seen, with arrangements of the cells into cords and pseudo-tubules. Leydig-like cells with vacuoles or a foamy cytoplasm were also present. Frozen sections stained with Sudan '.:\ showed the presence of fat in the Leydig cells. DISCUSSION
The androblastoma was first introduced by Teilum 1 for tumors of the testicle homologous to arrhenoblastomas (Meyer) 2 of the ovary. According to Friedman and l\1-oore, 3 they constitute 0.4 per cent of testicular tumors. are tumors originating from a testicular blastoma in which a differentiation 1 Teilum, G.: Arrhenoblastoma~androblastoma; homologous ovarian and testicular tumors; including the HO-called "luteomas" and "adrenal tumors" of ovary and interstitial cell tumors of testis. Acta Path. et JVIicrobiol. Scandinav., 23: 252--64, 1946. 2 Meyer, R.: Tubulare (testiculare) und solide -Formen der Andreiblastoma ovarii und ihre Beziehung zur Vermiinnlichung. Beitr. z. Path. Anat. u.z. a.Ilg. Path., 84: 48,5--520, 1930. "Friedman, N. B. and :VIoore, R. A.: Tumors of t,he testis. A report of fl22 cases. Military Surg., 99: 573-93, lfl46.
862
AZIZ FAM A:"
ISHAK
tendency in the direction of Sertoli and/ or Leydig cells may take place.1· 4 Accordingly, these tumors are classified histologically into: 1) tubular (tubular adenoma of Pick) 5 ; 2) mixed, and 3) diffuse stromal types. The reader is referred to the excellent account of the histopathology of these tumors by Dixon and Moore. 6 To our knowledge only six diffuse and mixed androblastomas, three of Teilum,1 · 4 and three of Freidman and JVIoore,3 have been reported, to which cases we are adding a seventh. The age incidence of the published cases varied from 4 to 52 years. Our case of an infant aged 3½ months is the youngest so far reported. The arrhenoblastomas of the ovary produce virilizing effects in females, while the homologous androblastomas in the male are sometimes associated with feminizing effects. According to Teilum,1· 4 · 7 this apparently paradoxical behavior depends on the predominance of either lipoidal androgen-producing Leydig cells (stromal element), or of estrogen-producing Sertoli cells (tubular epithelial element). Of the six reported cases referred to above, only one (Teilum's) was associated with hormonal effects, viz., gynecomastia. Our patient did not show any signs of hormonal disturbance, though admittedly, hormonal assays were not performed. None of the reported cases was malignant, although as Dixon and Moore 6 point out, the possibility exists of malignant variants of androblastoma, as indicated by the not infrequent occurrence of malignant arrhenoblastomas. SUMMARY
The history and clinical picture of a case of androblastoma of the left testis in an infant 33'1 months old are described. The tumor presented as a painless swelling of the left side of the scrotum of 1 month's duration prior to admission. Signs of hormonal imbalance were absent. A left orchiectomy was performed, the child remaining alive and well until the present time. The gross and microscopic findings are described. The tumor was of the mixed type of androblastoma, with the presence of both tubular and diffuse stromal elements. A brief comment is made on the reported cases of androblastoma in the literature. 4 Teilum, G.: Estrogen-producing Sertoli cell tumors (androblastoma tubulare lipoides) of the human testis and ovary. Homologous ovarian and testicular tumors. III. J. Clin. Endocrinol., 9: 301-18, 1949. 5 Pick, L.: Ueber Neubildungen am Genitale bei Zwittern nebst Beitragen zur Lehre von den Adenomen des Hodens und Eierstockes. Arch. f. Gyntk., 76: 191-281, 1905. 6 Dixon, F. J. and Moore, R. A.: Tumors of the male sex organs, in Atlas of Tumor Pathology, Sect. VIII~Fasc. 31b & 32, pp. 120-126, Washington D. C.: Armed Forces Institute of Pathology, 1952. 7 Teilum, G.: Endocrine varieties of "androblastoma" in ovary and testis. J. Obst. & Gynaec. Brit. EmD .. 58: 201-2, 1951.