630
Brief clinical and laboratory observations
DISCUSSION The entity, inflammatory pseudotumor, also described as plasma cell granuloma,' fibroxanthomaJ and histiocytoma,' is an uncommon benign lesion in childhood. It occurs more frequently in adults. Most of the examples reported in the literature have been in the lung and upper respiratory tract.' According to Bahodori and Liebow,' plasma cell granuloma represents localized proliferations, predominantly of mature plasma cells, with Russell bodies, reticuloendothelial cells, and intermediate forms, supported by a stroma of granulation tissue. Other cellular elements, including lymphocytes and large mononuclear ceils, may co-exist with the plasma cells. The stroma may contain interlacing or whorled masses o f fibroblasts and may be focally ossified or calcified. It is often hyalinized, with an appearance similar to that of paramyloid. Most of the plasma cell granulomas described in children have been reported in the lung. ~ An intraabdominal inflammatory pseudotumor in a 14-year-old girl and a retroperitoneal inflammatory pseudotumor in a 60-year-old woman have been reported? However, the significance of a previous history of abdominal surgery was emphasized. There have been sporadic cases of extrapulmonic sites, including the stomach,' renal pelvis) liver,6 and an intrarenal mass lesion,' all occurring in adults. A similar syndrome consisting of fever, anemia, and hyperglobulinemia has been described in association with giant lymph node hyperplasia of plasma cell type.'. 9 Once again, these lesions occur most frequently in the mediastinum, but may occur wherever lymph nodes are found. Mesenteric involvement has been reported in three teenagers and one adult. Histologically this entity.is distinctive from plasma cell pseudotumor in that the lesions are altered lymph nodes. They are characterized by the presence of sheets of mature plasma cells in the interfol-
The Journal of Pediatrics October 1978
licular tissue, usually with normal to large-sized follicle centers. In most of the examples reported, multiple discrete lymph nodes comprised the clinically observed mass? In our patient, on the basis o f fever, hypergammaglobulinemia, increased plasma cells in the bone marrow, thrombocytosis, microcytic hypochromic anemia, and elevated haptoglobin, a preoperative diagnosis of chronic inflammatory disease was suspected. Unlike the two other patients with inflammatory pseudotumor of the abdomen, this child had no previous history of abdominal surgery. The etiology of this inflammatory mass remains uncertain. REFERENCES
I. Bahadori M, and Liebow AA: Plasma cell granulomas of the lung, Cancer 31:191, 1973. 2. Kauffman SL and Stout AP: ttistiocytic tumors (fibrous xanthoma and histiocytoma) in children, Cancer 14:469, 1961. 3. Wu JP, Unis EJ, Fetterman (3, Jaeschhe WF, and Gilbert EF: Inflammatory pseudotumor of the abdomen: plasma cell g~'anuloma, J Clin Pathol 26:943, 1973. 4. Soga J, Sarto K, Suzuki N, and Sakai T: Plasma cell granuloma of the stomach: a report of a case and review of the literature, Cancer 25:618, 1970. 5. Davides KC, Johnson SH III, Marshall M Jr, Price SE Jr, and Stravrides A: Plasma cell granuloma of the renal pelvis. J Urol 107:938, 1972. 6. Pack GT, and Baker HW: Total right hepatic lobectomy: report of a case, Am Surg 138:253, 1953. 7. Fisch AE, and Bradey PA: Plasma cell granuloma of kidney, Urology 8:89, 1976. 8. Keller AR, Hochholzer L, and Castleman B: ttyalinevascular and plasma-cell types of giant lymph node hyperplasia of the mediastinum and other locations, Cancer 29:670, 1972. 9. Kahn LB, Ranchod M, Stables DP, King tI, and Yuckelman I: Giant lymph node hyperplasia with haematological abnormalities, S Aft Med 47:811, 1973.
hCG stimulation test in prepttbertal boys with cryptorchidism Charalabos Anoussakis, M.D., Demosthenes Alexiou, M.D.,* Demetrios LiaRakos, M.D., and Pelros Skopelitis, Ch.D., Athens, Greece
THE ENDOCRINE TESTICULAR FUNCTION in children with undescended testes has been studied extensively *Reprint address: 2nd Department of Pediatrics, University of Athens, "'Aglaia K)'riakou" Children's Hospital, Athens, 617
Greece.
but the results show great discrepancies. Some investigators have found normal testicular function after stimulation of the testes) e hut diminished secretion of testosterone in 30 to 43% of cryptorchid patients has also been reported. '-9
0022-3476/78/100630+03500.30/0 9 1978 The C. V. Mosby Co.
l'oh,ne 93 Number 4
Brief clinical and laboratory observations
We studied the endocrine response of the testes to human chorionic gonadotropin stimulation in prepubertal boys with bilateral and unilateral cryptorchidism.
Table I. Mean plasma testosterone levels (ng/dl) before and after hCG stimulation of the testes in patients and controls
I
Abbreviation used hCG: human chorionic gonadotropin
Group MATERIALS
AND METItODS
The studied patients included 35 children with true cryptorchidism and 18 normal boys who served as controls. Both patients and controls were in the prepubertal stage of sexual development (Stage I of Tanner) and had normal height and weight. They also had normal bone ages according to the charts of Greulich and Pyle. The subjects were divided into the following study groups: I. Controls: There were 18 normal boys, 2V12 to 10"-/, years of age, with a mean of 51~ years. Eight of them were younger than 6 years (mean age 42/,2) and 10 were over 6 years (mean age 82/,2). II. Patients with bilateral cryptorchidism: There were 17 patients 3'/12 to 11 years of age, with a mean of 71/12 years. Seven of them were younger than 6 years (mean age 4'/,2) and the rest were over 6 years (mean age 8V,2). All patients h/ld true undescended testes in the inguinal canal except one with complete cryptorchidism. III. Patients with unilateral cryptorchidism: There were 18 patients, 3'/,2 to 1 IV, years o f a g e with a mean of 7VI_~ years. Seven of them were younger than 6 years (mean age 4V,) and 11 were over 6 years (mean age 9"-/1.-). All patients had a unilaterally undescended testis except one with complete unilateral cryptorchidism. Prolonge d stimulation of the testes by hCG resulted in increased secretion of testosterone? Thus, cryptorchid patients who had beim given hCG during the last six months were excluded from the study. We also excluded patients operated for cryptorchidism, boys with retractile testes, as weU as anorchic and obese patients. In both patients and controls the plasma testosterone 9level was measured before and after hCG stimulation, which was performed as follows: three chorionic gonadotropin injections, 1,500 IU each, were given daily. Blood 99 were obtained in the morning (10 to 12 Ara) before the first injection and 24 hours after the last one. The plasma, after separation, was kept at - 2 0 ~ The determination of plasma testosterone was made by radioimmunoassay. RESULTS The age differences among the studied groups were not statistically significant (P > 0.05). The mean plasma testosterone values, before and after hCG stimulation, are
63 1
Controls Bilateral cryptorchidism Unilateral cryptorchidism
Plasma testosterone
No. of ] Before hCG [ After hCG patients ](,,,ean • SD) I(n,ean • SD) 18 17
14.7 • 5.5 14.0 • 7.6
166.2• 36.2 130.5__. 49.1
18
16.6 ___6.2
172.7• 43.2
Table II. Mean plasma testosterone levels (ng/dl) before and after hCG stimulation of the testes according to age
Plasma testosterone Gro,tps
No. of patients.
Controls Age <_ 6 yr 8 Age > 6 yr 10 9Bilateral cryptorchidism Age _< 6 yr 7 Age > 6 yr 10 Unilateral cryptorchidism Age _< 6 yr 7 Age > 6 yr II
BeforehCG ] After hCG (mean • SD) I (mean • SD) 10.3 _ 3.15 18.3 _+ 4.2
164.7___33.6 167.4 • 39.9
10.6 -4- 2.9 17.7 • 8.6
161.8 ___42.3 108.6 • 42.3
13.3 • 6.2 18.7 • 5.2
189.4 • 42.8 162.1 • 4i.9
seen in Table I. The differences between patients with bilateral cryptorchidism and controls were not statistically significant before stimulation (P > 0.05), but the differences were significant following stimulation (t = 2.457; P < 0.05). The differences betwee n unilateral cryptorchid patients and the normal boys before and after hCG stimulation were not statistically significant (P > 0.05). After the age of 6 years there was a sharp decline of testosterone secretion in the group of patients with bilateral cryptorchidism. Thus, we divided each of the studied groups into two subgroups (Table ll). The differences between the normal boys under 6 years and the patients of corresponding age with bilateral or unilateral cryptorchidism, before as well as after hCG stimulation, were not statistically significant (P > 0.05). The only statistically significant difference (t = 3,192; P < 0.01) was found between the plasma testosterone levels of the normal boys above 6 years of age and the corresponding patients wlth bilateral cryptorchidism. DISCUSSION It has been stated that the endocrine function of the testes of cryptorchid patients is normal s.6 or satisfac-
63 2
Brief clinical and laboratory observations
tory?" Others have reported a decreased response of the testes in some of their patients with cryptorchidism?- ~ These contradictory results could be attributed to the small number of patients studied,-". 8. ,o to the differing d0ses ofthe hCG administered," ~. 5as well as to improper grouping of the subjects, i.e., bilateral and unilateral cryptorchid patients, 8''' ,o or operated and non-operated patients with cryptorchidism~"9 Were studied together. In the present investigation all conditions that could - affect testicular function were excluded. Our results indicate that in patients with bilateral cryptorchidism there is a diminution of testosterone secretion after the age of 6 years. This suggests that time is an important factor in the treatment ofcryptorchidism; impaire d endocrine function of the testes can be found after the age of 6 years, as has been observed for spermatogenesis. REFERENCES
1. Rivarola MA, Bergada C, and Gullen M: HCG stimulation test in prepubertal boys with cryptorchidism, in bilateral anorchia and in male pseudohermaphroditism, J Clin Endocrinol 31:526, 1970. 2. Levine LS, and New MI: Preoperative detection of hidden testes, Am J Dis Child 121:176, 1971. 3. Winter JSD,Taraska S, and Faiman C: The hormonal response to HCG stimulation in male children and adolescents, J Clin Endocrinol 34:348, 1972.
The Journal of Pediatrics October 1978
4. Sizonenco PC, Cuendet A, and Paunier L: FSH. 1. Evidence for its mediating role on testosterone secretion in cryptorchidism, J Clin Endocrinol Metab 37:68, 1973. 5. Bardin CW, Ross GT, Rifkind AB, Cargille CM, and Linsett MB: Sttidy of the pituitary-Leydig cell axis in young men with hypogonadotropic hypogonad!sm and hyposmia: Comparison w!th normal men, prepubertal boys and hypopituitary patients, J Clin Invest 48:2046, 1969. 6. Perheentupa J, Dessypris A, and Adlercreutz N: Plasma testosterone levels in boys following gonadotropin stimulation Acta PaediatrScand 61:265, 1972. 7. Rudd BT, Rayner RHW, Smith MR, ttolder G, Jivani SKM, and Theodoridis CG: Effect 6f human chorionic gonadotropin on plasma and urine testosterone in boys with delayed puberty, Arch Dis Child 48:590, 1973. 8. Canlorbe P, Toublanc JE, Job JC, Scholler R, Roger M, and Castanier M: Fonction endocrine du testicule dans 125.cas de cryptorchidies, Ann Endocrinol (Paris) 35:177, 1974. 9. Canlorbe P, Toublanc JE, Job JC, SchoUer R, Roger M, Castanier M, and Leymarie P: La fonction endocrine du testicule chez renfant et l'adolescent, Ann Pediatr 21:13, 1974. 10. Cacciari E, Cicognani A, Tasson! P, Flamigni P, Bolelli F, Pirazzoli P, and Lalardi S: Plasma testosterone and estradiol concentration in prepubertal boys with cryptorchidism before and after dexamethazone and after human chorionic g0nadotropin administration, Helv Paediatr Acta 29:27, 1974.
Response to influenza virus vaccine in children receiving chemotherapy.for malignancy William A. Smithson, M.D.,* Robert A. Siem, Ph.D., Roy E. Ritts, Jr., M.D., Gerald S. Gilchrlst, M.B., B,Ch., E. Omer Burgert, Jr., M.D., Duane M. ilstrup, M.S., and Thomas F. Smith, Ph.D., Rochester, Minn.
PARTICIPATING in a program to immunize high-risk groups against a new influenza serotype A / N e w Jersey/ 76 ("swine flu"),' we immunized 27 children who had malignant disease. Because earlier reports had indicated From the Ma)'o Medical Schoo# the Departments of Pediatrics, Microbiolog), and Laboratory Medichw, Ma)'o Clinic and Ma)'o Foundation; and the Section of Virus and Rickettsia, Minnesota Department of Health. Supported in part b)' Research Grant CA-4646"from the National Institutes of Health. Public Health Service, and b), a grant from the Pardee Foundation. *Reprint address: Departmentof Pediatrics, Mayo Clinic. Rochester, MN 55901.
diminished antibody response in this immunosuppressed group, ~ we measured the antibody response to the vaccine. Since the immunization program ceased before completion, only ten of the 2 7 patients received tl~e recommended two-dose course? Abbreviations used CCA: chick-cell agglutinating IIAI: hemagglutinin-inhibiting The patients were children whose ages ranged from 2 to 15 (mean 8.2) years; t h e r e were 13 boys and 14 girls. Eighteen had acute leukemia, four had lymphoma, and five had solid tumors. All but one patient with stable
0022-3476/78/100632+03500.30/0 9 1978 The C. V. Mosby Co.