ABSTRACT
Implant of artificial penile nodule (APN) is a socio-cultural practice, linked to penitentiary environment in French Guiana. Physicians are often unfamiliar with its existence. Although serious complications remain low regarding the high prevalence of this practice, urgent cares could be required. Indeed, implant of nodule can have functional sequelae, and sometimes life-threatening consequences, especially if infection occurs and spreads. We have reported the case of a 23-year-old male who presented an infection of the penis after the implant of two APN. Removal of the nodules associated with oral antibiotics was needed. We also present CT-scan images of another patient, as an example of fortuitous discovery of these nodules. We finally discuss the various complications already described in literature.
Le port de nodules péniens artificiels (NPA) est fortement lié à la fréquentation du milieu carcéral en Guyane française. Cette pratique est peu connue des professionnels de santé. Bien que les complications restent peu fréquentes malgré la prévalence élevée de ces nodules dans certaines populations, elles peuvent nécessiter une prise en charge diagnostique et thérapeutique urgente. En effet, il existe des risques fonctionnels, mais également vitaux survenant dans les suites d'une complication notamment infectieuse. Nous rapportons ici le cas d'une infection de la verge suite à l'implant de deux NPA chez un patient de 23 ans, pour laquelle le retrait des nodules et une antibiothérapie orale ont été nécessaires. Preuve de la forte prévalence de cette pratique, nous illustrons à l'aide d'une iconographie radiologique originale le cas d'un autre patient chez qui ces nodules ont été fortuitement découverts. Enfin, nous discutons des différentes complications décrites dans la littérature.
Subject(s)
Penile Diseases/diagnosis , Penile Prosthesis/adverse effects , Prosthesis-Related Infections/diagnosis , Administration, Oral , Anti-Bacterial Agents/administration & dosage , Device Removal , French Guiana , Humans , Male , Penile Diseases/drug therapy , Penile Diseases/surgery , Prisons , Prosthesis Design/adverse effects , Prosthesis-Related Infections/drug therapy , Prosthesis-Related Infections/surgery , Risk Factors , Young AdultABSTRACT
Lymphedema is the result of an alteration of the lymphatic drainage, and its most common worldwide cause is filariasis. In our practice usually is associated to neoplasic, inflammatory and granulomatous processes, radiotherapy, hydroelectrolytic disbalances, and idiopathic. It can affect any part of the body, including the penis and scrotum. The genital lymphedema is a rare presentation, it corresponds to 0.6% of lymphedema. However, causes serious functional, social and emotional limitations for the patient. Too often have pain, recurrent infections, sexual dysfunction, cosmetic deformity, sometimes it limits mobility and ambulation. Although there are several treatment options, both medical and surgical, it has not been found ideal for this disease. We present a 43 years old patient with penoscrotal lymphedema due to hidradenitis suppurativa, it limits his normal activity. The patient was referred to our center after unsuccessful medical treatment (doxycycline and clindamycin cycles). Surgical treatment consisted of total excision of the skin and subcutaneous tissue to Buck's fascia. Split thickness skin grafts were used to cover the defect. The result was satisfactory both functionally and aesthetically.
El linfedema es producto de una alteración en el drenaje linfático, y su causa más frecuente en todo el mundo es la filariasis. En nuestro medio suele encontrarse asociada a procesos neoplásicos, inflamatorios, granulomatosos, secuelas por radioterapia, desequilibrios hidroelectrolíticos y procesos idiopáticos. Puede afectar a cualquier parte del cuerpo, incluyendo el pene y el escroto. El linfedema genital es una presentación infrecuente, que corresponde al 0.6% de los linfedemas. No obstante, causa graves limitaciones funcionales, sociales y emocionales para el paciente. Con mucha frecuencia se presentan dolor, infecciones recurrentes, disfunción sexual y deformidad estética, llegando incluso a limitar la movilidad y la deambulación. Aunque existen varias opciones de tratamiento, tanto médico como quirúrgico, no se ha encontrado el ideal para esta enfermedad. Presentamos el caso de un paciente de 43 años afecto de hidrosadenitis axilar e inguinal que padece linfedema penoescrotal grave que limita seriamente su actividad habitual. El paciente fue remitido a nuestro centro tras el fracaso del tratamiento con antibióticos (ciclos de doxiciclina y clindamicina). El tratamiento quirúrgico consistió en la resección de piel y tejido celular subcutáneo hasta fascia de Buck y cobertura con injertos de piel. El resultado final fue adecuado desde un punto de vista tanto funcional como estético.
Subject(s)
Genital Diseases, Male/etiology , Hidradenitis Suppurativa/complications , Lymphedema/etiology , Scrotum , Adult , Clindamycin/therapeutic use , Cutaneous Fistula/etiology , Doxycycline/therapeutic use , Genital Diseases, Male/diagnostic imaging , Genital Diseases, Male/drug therapy , Genital Diseases, Male/surgery , Hidradenitis Suppurativa/drug therapy , Hidradenitis Suppurativa/surgery , Humans , Lymphedema/surgery , Magnetic Resonance Imaging , Male , Penile Diseases/diagnostic imaging , Penile Diseases/drug therapy , Penile Diseases/etiology , Penile Diseases/surgery , Scrotum/surgery , Skin TransplantationABSTRACT
A 70-year-old male presenting a 3-month history of genital painless erythematous nodules in the balanopreputial sulcus was referred to our service. Histopathological exam presented a chronic dermatitis with epithelioid granulomas and Grocott staining revealed numerous fungal structures with a suggestive morphology of Histoplasma sp. Cultures evidenced Histoplasma capsulatum var. capsulatum. Treatment with oral itraconazole led to complete remission of lesions.
Subject(s)
Dermatomycoses/pathology , Histoplasmosis/pathology , Immunocompetence , Penile Diseases/pathology , Aged , Antifungal Agents/therapeutic use , Biopsy , Dermatomycoses/drug therapy , Dermatomycoses/immunology , Histoplasma , Histoplasmosis/drug therapy , Histoplasmosis/immunology , Humans , Immunocompetence/immunology , Itraconazole/therapeutic use , Male , Penile Diseases/drug therapy , Penile Diseases/immunology , Treatment OutcomeABSTRACT
A 70-year-old male presenting a 3-month history of genital painless erythematous nodules in the balanopreputial sulcus was referred to our service. Histopathological exam presented a chronic dermatitis with epithelioid granulomas and Grocott staining revealed numerous fungal structures with a suggestive morphology of Histoplasma sp. Cultures evidenced Histoplasma capsulatum var. capsulatum. Treatment with oral itraconazole led to complete remission of lesions.
Subject(s)
Aged , Humans , Male , Dermatomycoses/pathology , Histoplasmosis/pathology , Immunocompetence , Penile Diseases/pathology , Antifungal Agents/therapeutic use , Biopsy , Dermatomycoses/drug therapy , Dermatomycoses/immunology , Histoplasma , Histoplasmosis/drug therapy , Histoplasmosis/immunology , Immunocompetence/immunology , Itraconazole/therapeutic use , Penile Diseases/drug therapy , Penile Diseases/immunology , Treatment OutcomeABSTRACT
Herpes zoster is a common dermatological condition which affects up to 20 percent of the population, most frequently involving the thoracic and facial dermatomes with sacral lesions occurring rarely and only a few reported cases of penile shingles. Case report: We report two cases of unusual penile clinical presentations of varicella zoster virus infection in immunocompetent men. The patients presented with grouped clusters of vesicles and erythema on the left side of penile shaft and posterior aspect of the left thigh and buttock, involving s2-s4 dermatomes. The lesions resolved quickly upon administration of oral antiviral therapy. Conclusion: Penile herpes zoster should not be overlooked in patients with unilateral vesicular rash.
Subject(s)
Adult , Aged , Humans , Male , Herpes Zoster/pathology , Penile Diseases/pathology , Acyclovir/therapeutic use , Antiviral Agents/therapeutic use , Buttocks , Herpes Zoster/drug therapy , Penile Diseases/drug therapy , Penile Diseases/virology , Skin Diseases, Viral/drug therapy , ThighABSTRACT
UNLABELLED: Herpes zoster is a common dermatological condition which affects up to 20% of the population, most frequently involving the thoracic and facial dermatomes with sacral lesions occurring rarely and only a few reported cases of penile shingles. CASE REPORT: We report two cases of unusual penile clinical presentations of varicella zoster virus infection in immunocompetent men. The patients presented with grouped clusters of vesicles and erythema on the left side of penile shaft and posterior aspect of the left thigh and buttock, involving s2-s4 dermatomes. The lesions resolved quickly upon administration of oral antiviral therapy. CONCLUSION: Penile herpes zoster should not be overlooked in patients with unilateral vesicular rash.
Subject(s)
Herpes Zoster/pathology , Penile Diseases/pathology , Acyclovir/therapeutic use , Adult , Aged , Antiviral Agents/therapeutic use , Buttocks , Herpes Zoster/drug therapy , Humans , Male , Penile Diseases/drug therapy , Penile Diseases/virology , Skin Diseases, Viral/drug therapy , ThighABSTRACT
INTRODUCTION: Despite the fact that there is minimal evidence-based data supporting it, the concept of pharmacological penile rehabilitation following radical prostatectomy (RP) is receiving great attention. AIM: To define attitudes and practice patterns of clinicians who were members of the International Society for Sexual Medicine (ISSM) and/or its affiliated societies. METHODS: Members of the ISSM and its regional affiliates were invited to participate in a web-based survey. MAIN OUTCOME MEASURES: Demographic factors, current practice status, and opinions regarding post-RP erectile dysfunction and penile rehabilitation. The statistical methods used included chi-square, Student's t-tests, and logistic regression analysis. RESULTS: Three hundred-one physicians from 41 countries completed the questionnaire (82% were urologists). Sixty-five percent of the responders had formal sexual medicine specialty training, 44% had uro-oncology specialty training, and 60% performed RPs. Eighty-seven percent performed some form of rehabilitation. As part of the primary rehabilitation strategy, 95% used phosphodiesterase type 5 inhibitors (PDE5), 30% used vacuum device, 75% used intracavernosal injections, and 9.9% used intraurethral prostaglandin. Fifty-four percent commenced rehabilitation immediately/just after urethral catheter removal, and 37% within the first 4 months after RP. Neither the number of years in medical practice, clinician age, nor country/region of practice differed between rehabilitation performers and non-performers. With regard to the primary reason for avoiding rehabilitation: 50% responded said it is the cost; 25% said the fact that it is not evidence-based; and 25% said they were not familiar with the concept. Performing rehabilitation was positively associated with urologic oncology training (P = 0.03), performing RP (P < 0.001), and seeing over 50 post-RP patients per year (P = 0.011). CONCLUSIONS: Among ISSM members post-RP penile rehabilitation is widely practiced, commenced early, and based predominantly on PDE5 inhibitors and intracavernosal injections. Clinicians who perform RP or see over 50 such patients per year are the most likely to perform rehabilitation. Cost represents the most common reason for rehabilitation neglect.
Subject(s)
Impotence, Vasculogenic/drug therapy , Penile Diseases/drug therapy , Penis/drug effects , Phosphodiesterase Inhibitors/therapeutic use , Piperazines/therapeutic use , Practice Patterns, Physicians'/statistics & numerical data , Prostatectomy/adverse effects , Sulfones/therapeutic use , Aged , Confidence Intervals , Data Collection , Health Knowledge, Attitudes, Practice , Humans , Impotence, Vasculogenic/rehabilitation , Logistic Models , Male , Middle Aged , Odds Ratio , Penile Diseases/rehabilitation , Purines/therapeutic use , Sildenafil Citrate , Surveys and Questionnaires , Time FactorsABSTRACT
BACKGROUND: Histoplasma capsulatum is the causative agent of American histoplasmosis. The relationship between disseminated histoplasmosis and AIDS has been well established. Widespread hematogenous dissemination of Histoplasma capsulatum in HIV positive patients can cause a plethora of clinical manifestations; virtually any organic system can be affected. However, genital ulceration by H. capsulatum in patients with AIDS is a real challenge during investigation of the infection due to the great variety of differential diagnoses that are involved. METHOD: The diagnosis was performed by histopathologic study; H. capsulatum was detected by silver staining (Grocott staining) and confirmed by immunocytochemistry. RESULTS: We report three cases of histoplasmosis in patients with AIDS, in which we observed genital ulcers, an unusual form of presentation of this disease. In one of these cases, the treatment resulted in total cure. CONCLUSION: The cases reported herein are to illustrate the plurality of pathologies and clinical manifestations, which may affect immunocompromised patients. The correct diagnosis of fungal diseases in these patients following well established treatment will improve the prognosis.
Subject(s)
AIDS-Related Opportunistic Infections/microbiology , Histoplasma/isolation & purification , Histoplasmosis/microbiology , Penile Diseases/microbiology , Skin Ulcer/microbiology , AIDS-Related Opportunistic Infections/pathology , Adult , Antifungal Agents/therapeutic use , Fatal Outcome , Histoplasmosis/drug therapy , Histoplasmosis/pathology , Histoplasmosis/virology , Humans , Male , Penile Diseases/drug therapy , Penile Diseases/pathology , Penile Diseases/virology , Skin Ulcer/drug therapy , Skin Ulcer/pathology , Skin Ulcer/virologyABSTRACT
Ten patients with donovanosis were treated with thiamphenicol for two weeks. In eight of them, included two HIV infected patients, lesions healed. The safety profile of thiamphenicol makes it a useful and cost-effective agent in the management of donovanosis. Randomized controlled trials should be conducted with these treatment options.
Subject(s)
Anti-Bacterial Agents/therapeutic use , Granuloma Inguinale/drug therapy , Penile Diseases/drug therapy , Thiamphenicol/therapeutic use , Follow-Up Studies , Humans , Male , Penile Diseases/microbiology , Treatment OutcomeABSTRACT
Ten patients with donovanosis were treated with thiamphenicol for two weeks. In eight of them, included two HIV infected patients, lesions healed. The safety profile of thiamphenicol makes it a useful and cost-effective agent in the management of donovanosis. Randomized controlled trials should be conducted with these treatment options.
Subject(s)
Humans , Male , Anti-Bacterial Agents/therapeutic use , Granuloma Inguinale/drug therapy , Penile Diseases/drug therapy , Thiamphenicol/therapeutic use , Follow-Up Studies , Penile Diseases/microbiology , Treatment OutcomeABSTRACT
Two mucocutaneous leishmaniasis cases resistant to therapy are reported here. After the failure of initial therapies (antimony, amphotericin B and/or pentamidine) patients received a low-dose schedule: one ampoule of meglumine antimoniate (405 mg of pentavalent antimony [Sb v]) by intramuscular injection, three times a week until complete healing of the lesions. One patient was cured with a total of 30 ampoules in 10 weeks and the other received 36 ampoules in 12 weeks. Both remain clinically cured after one year of follow-up.
Subject(s)
Antiprotozoal Agents/administration & dosage , Leishmaniasis, Cutaneous/drug therapy , Meglumine/administration & dosage , Organometallic Compounds/administration & dosage , Adult , Drug Administration Schedule , Ear Diseases/drug therapy , Ear, External , Follow-Up Studies , Humans , Injections, Intramuscular , Male , Meglumine Antimoniate , Middle Aged , Penile Diseases/drug therapy , Treatment OutcomeABSTRACT
OBJECTIVE: To analyze the clinical and histological findings in boys with bilateral anorchia and the response to testosterone treatment on penis length. STUDY DESIGN: Patients were divided into two groups according to the absence (group A, n = 29) or the presence (group B, n = 26) of palpable intrascrotal or inguinal mass at first clinical examination. RESULTS: A micropenis was found in 46% of patients (n = 24) with a similar proportion in both groups. Testosterone treatment induced a mean penis length gain of 1.9 +/- 1.3 SDS (standard deviation score). However, micropenis persisted in six patients. Histological examination (n = 18) confirmed the absence of any testicular structure with deferent ducts being present unilaterally or bilaterally in all but three patients. In these three patients, a hemorrhagic testis, probably as a result of a mechanical torsion, was found. CONCLUSIONS: The presence of isolated micropenis in almost half of patients with bilateral anorchia strongly suggests that the testicular damage frequently occurs during the second half of gestation after male sexual differentiation. In most cases, testosterone treatment stimulates the penile growth. Although the pathogenesis of bilateral anorchia may be heterogeneous, our study suggests that gonads may have been functionally abnormal before they disappeared, and suggests that some patients have an intrinsic endocrine disorder.
Subject(s)
Androgens/therapeutic use , Eunuchism/drug therapy , Eunuchism/pathology , Penile Diseases/drug therapy , Penile Diseases/pathology , Testosterone/therapeutic use , Androgens/deficiency , Child , Child, Preschool , Eunuchism/congenital , Eunuchism/surgery , France/epidemiology , Genitalia, Male/abnormalities , Genitalia, Male/drug effects , Genitalia, Male/growth & development , Genitalia, Male/surgery , Humans , Infant , Infant, Newborn , Male , Penile Diseases/congenital , Penile Diseases/surgery , Spermatic Cord Torsion/congenital , Spermatic Cord Torsion/pathology , Spermatic Cord Torsion/surgery , Testosterone/deficiency , Time Factors , Treatment Outcome , Urologic Surgical Procedures, MaleABSTRACT
OBJECTIVE: To compare clinical improvement between treatment with metilprednisolone aceponate vs. betamethasone dipropionate among children with nonretractable prepuce. MATERIAL AND METHODS: Between August 2001 and November 2002, we carried out a double blind and controlled clinical trial in 34 children with a diagnosis of nonretracable prepuce. Children were randomly assigned to one of the following groups and topical treatment was administered: Group A; metilprednisolone aceponate 0. I 1% and Group B; betamethasone dipropionate 0.05%. RESULTS: Improvement was noted in 88.2% of our sample studied; (n= 15) children from group A and 76.4% (n= 13) childrenfrom group B; however, we did not observe a significant difference when comparing percentages between the two groups (chi2 = 0.2; p = 0.6). CONCLUSIONS: The percentage of clinical improvement was similar between the two groups of topical steroid treatment administered.
Subject(s)
Betamethasone/analogs & derivatives , Glucocorticoids/administration & dosage , Methylprednisolone/analogs & derivatives , Penile Diseases/drug therapy , Adolescent , Betamethasone/administration & dosage , Child , Child, Preschool , Double-Blind Method , Humans , Male , Methylprednisolone/administration & dosage , Remission InductionABSTRACT
Objetivo: Comparar el porcentaje de mejoría clínica entre aceponato de metilprednisolona versus dipropionato de betametasona tópicos, en niños con prepucio no retráctil. Material y métodos: De agosto del 2001 a noviembre de 2002 se realizó un estudio clínico, doble ciego y controlado en 34 niños con diagnóstico de prepucio no retráctil. Los niños fueron asignados al azar en los siguientes grupos de tratamiento tópico: grupo A; aceponato de metilprednisolona 0.1% y grupo B; dipropionato de betametasona a 0.05%. Resultados: De los 34 pacientes analizados se obtuvo mejoría en 88.2% (n = 15) del grupo A y 76.4% (n = 13) del grupo B, sin embargo, no hubo diferencia significativa en la comparación de porcentajes entre los dos grupos estudiados (χ2 = 0.2; p = 0.6). Conclusiones: El porcentaje de mejoría clínica entre los dos tratamientos de esteroides tópicos fue semejante.
OBJECTIVE: To compare clinical improvement between treatment with metilprednisolone aceponate vs. betamethasone dipropionate among children with nonretractable prepuce. MATERIAL AND METHODS: Between August 2001 and November 2002, we carried out a double blind and controlled clinical trial in 34 children with a diagnosis of nonretracable prepuce. Children were randomly assigned to one of the following groups and topical treatment was administered: Group A; metilprednisolone aceponate 0. I 1% and Group B; betamethasone dipropionate 0.05%. RESULTS: Improvement was noted in 88.2% of our sample studied; (n= 15) children from group A and 76.4% (n= 13) childrenfrom group B; however, we did not observe a significant difference when comparing percentages between the two groups (chi2 = 0.2; p = 0.6). CONCLUSIONS: The percentage of clinical improvement was similar between the two groups of topical steroid treatment administered.
Subject(s)
Humans , Male , Child, Preschool , Child , Adolescent , Betamethasone/analogs & derivatives , Penile Diseases/drug therapy , Glucocorticoids/administration & dosage , Methylprednisolone/analogs & derivatives , Betamethasone/administration & dosage , Double-Blind Method , Methylprednisolone/administration & dosage , Remission InductionABSTRACT
OBJECTIVES: On December 1905 Darling, resident histopathologist on the Panama Channel area, observed a parasite in the large mononuclear cells of different organs. Negroni in 1914 described the first Argentinean case. Lung, oral, nasal, and laryngeal involvement are common. On the other hand, genital involvement is uncommon. The objective of this war is to communicate the case of a patient with genital histoplasmosis. METHODS/RESULTS: A 60-year-old Argentinian male started two years before with penile lesions and pruritus. Two partial penile resections had been performed in 1999, but pathology reports were not available. The lesions evolved towards ulceration and bleeding. Surgical toilette was carried out. A partial penectomy and glanuloplasty with an original technique were performed. Pathology revealed intracellular and extracellular spherical microorganisms. Morphologically it was histoplasma capsulatum. The histoplasmosis is an endemic mycosis. The infection takes place when inhaling the microconidiae of the fungus filamentous phase. Only a small number of people develop the disease after infection. Diagnosis is made by discovering of 2-3 micron yeasts with Giemsa stain. CONCLUSIONS: If a patient from the histoplasmosis endemic area has a granular lesion, a mycotic disease should be considered.
Subject(s)
Granuloma/diagnosis , Histoplasmosis/diagnosis , Penile Diseases/diagnosis , Antifungal Agents/therapeutic use , Argentina , Combined Modality Therapy , Granuloma/drug therapy , Granuloma/microbiology , Granuloma/surgery , Histoplasmosis/drug therapy , Histoplasmosis/surgery , Humans , Itraconazole/therapeutic use , Male , Middle Aged , Penile Diseases/drug therapy , Penile Diseases/microbiology , Penile Diseases/surgery , Staining and Labeling , Surgical FlapsABSTRACT
Two patients from the gold mines of Bolivar State, Venezuela, presenting cutaneous leishmaniasis in the genital region, an unusual location, are described. The first patient showed an ulcerated lesion of the glans penis. Leishmanin skin test was positive. A biopsy specimen revealed a granulomatous infiltrate containing Leishmania parasites. In the second patient, Leishmanin skin test was positive, HIV and VDRL were negative. Leishmania parasites were present in a biopsy of an ulcerated lesion in the scrotum, with an indurated base, infiltrative borders with an yellowish exudate. Patients were treated with meglumine antimoniate and the lesions healed.
Subject(s)
Leishmaniasis, Mucocutaneous , Penile Diseases/parasitology , Adolescent , Adult , Antiprotozoal Agents/therapeutic use , Humans , Leishmaniasis, Mucocutaneous/drug therapy , Male , Meglumine/therapeutic use , Meglumine Antimoniate , Organometallic Compounds/therapeutic use , Penile Diseases/drug therapyABSTRACT
O presente artigo trata do relato de um caso raro na clínica dermatológica, de um caminhoneiro com donovanose peniana, ulcerada, com fundo de aspecto framboesiforme, tratada inicialmente com a associação de sulfametoxazol/trimetoprima e depois com fosfato de tetraciclina com cura total das lesões sem deixar cicatrizes deformantes
Subject(s)
Humans , Male , Adult , Penile Diseases/diagnosis , Penile Diseases/drug therapy , Granuloma Inguinale/diagnosis , Granuloma Inguinale/drug therapyABSTRACT
A patient with generalized lichen planus with lesions in the infrequent localization like face, palms, soles and an extensive erosion of glans penis, is described in a 46 year old patient. He had received various treatment which include systemic steroids, without improvement. After treatment with thalidomide (initial doses 300 mg/day for 2 weeks and 200 mg/day for further 10 weeks) he presents resolution of his lesions and symptomatology. A review of lichen planus etiopathogenesis, making emphasis in the immunological hypothesis is made. As well as the different uses and action mechanism of thalidomide in various inflammatory dermatoses.
Subject(s)
Lichen Planus/drug therapy , Penile Diseases/drug therapy , Humans , Lichen Planus/pathology , Male , Middle Aged , Penile Diseases/pathologyABSTRACT
The association of hypoglycemia and microphallus in the male neonate is presumptive evidence of congenital hypopituitarism. This was observed in four male infants with normal birth weight and length, optic discs, and intelligence, and without gross central nervous system malformations. Plasma and urinary cortisol values were low. Stimulation with metyrapone and insulin hypoglycemia failed to elicit a rise in plasma corticoids, but multiple doses of ACTH evoked a response. Growth hormone responses to arginine, insulin, sleep, L-dopa, and glucagon were uniformly less than 2.5 ng/ml. In three patients, however, length remained within 2 SD of the mean until two years of age; in one, there was a sharp decrease in growth by three months. Two patients had low plasma TSH and thyroxine concentrations within the first month of life. In the other two patients, whose thyroxine levels were measurable, intravenous administration of thyrotropin-releasing factor evoked a normal rise in plasma TSH; serum thyroxine decreased into the hypothyroid range in one after GH therapy was initiated. Plasma prolactin was normal in the first two patients receiving thyroxine replacement therapy. The other two patients had elevated baseline prolactin levels and had an augmented rise in plasma prolactin after administration of TRF. Human chorionic gonadotropin induced a 10- to 15-fold rise in plasma testosterone in the two patients tested. The changes in plasma FSH and LH after luteinizing hormone-releasing factor were either low or in the prepubertal range. In three patients, treated with testosterone enanthate intramuscularly, phallic growth occurred. In addition, all three had a transient increase in height but no acceleration of skeletal maturation. The data suggest a deficiency of hypothalamic hypophysiotropic hormones rather than a primary pituitary defect. Early recognition of this syndrome complex is critical for prompt treatment of the life-threatening cortisol deficiency. The diagnosis is more difficult in affected females because their external genitals are normal. The microphallus is a remediable manifestation of hypopituitarism.