Mitotane treatment in patients with metastatic testicular Leydig cell tumor associated with severe androgen excess.

Mitotane (o,p'DDD) is established in the adjuvant and advanced-stage treatment of adrenocortical carcinoma and counteracts both tumor growth and tumor-related steroid production. Both the adrenal glands and the gonads are steroidogenically active organs and share a common embryogenic origin. Here, we describe the effects of mitotane in two patients with metastatic Leydig cell tumor (LCT) of the testes and associated severe androgen excess (serum testosterone 93 and 88 nmol/L, respectively; male reference range 7-27 nmol/L). Both men suffered from severe restlessness, insomnia and irritability, which they described as intolerable and disrupting normal life activities. Urinary steroid profiling by gas chromatography-mass spectrometry (GC-MS) confirmed excess androgen production and revealed concurrent overproduction of glucocorticoids and glucocorticoid precursors, which under physiological conditions are produced only by the adrenal glands but not by the gonads. In a palliative approach, they were commenced on mitotane, which achieved swift control of the hormone excess and the debilitating clinical symptoms, restoring normal quality of life. GC-MS demonstrated normalization of steroid production and decreased 5α-reductase activity, resulting in decreased androgen activation, and imaging demonstrated disease stabilization for 4-10 months. In conclusion, mitotane can be highly effective in controlling steroid excess in metastatic LCTs, with anti-tumor activity in some cases.

Mammary gland tumors in a male Cocker Spaniel.

BACKGROUND: Mammary gland tumors are the most common tumors in sexually intact female dogs; however, they are rare in male dogs. This study was conducted to investigate the relationship between sexual hormones and mammary gland tumors in a male dog. CASE PRESENTATION: A 13-year-old, intact male Cocker Spaniel presented to the Veterinary Teaching Hospital of Kangwon National University, Republic of Korea, with an acute right ruptured caudal abdominal mass. Physical examination revealed a 14 × 14 cm ruptured mass in the right caudal abdomen, as well as a 1.5 × 1.5 cm mass in the first right mammary gland. The estrogen and progesterone concentrations in serum were within normal levels. Total mastectomy was done on the right side mammary glands. Following surgery, the site was fully recovered; however, a mass that had grown to 2 × 2 cm was found in the left fifth mammary gland and a testis tumor was also found over the period of 4 months. Mastectomy was performed on the left caudal mammary gland and castration was also performed. After the final surgery, the dog fully recovered. Histopathological examination of all three masses revealed high grade mammary adenocarcinoma in the mammary gland and the testis was diagnosed as Leydig cell adenoma. Immunohistochemical analysis revealed that the estrogen and progesterone receptors were expressed on limited cells in mammary and testis tumors. CONCLUSION: The results of this study suggest that mammary tumors and testes tumors can occur in male dogs without relationship to female sexual hormone.

Leydig cell tumor with lung metastasis diagnosed by lung biopsy.

Leydig cell tumors are very rare and account for only 3% of testicular tumors and are generally benign. Only less than 0.2% of all testicular cancers were evidenced by metastatic spread. We report a 34-year-old man visited hospital because of coughing sputum mixed with blood. His chest CT showed bilateral patch clouding opacity. He was suspected with allergic alveolitis and treated with methylprednisolone. However, his symptoms and general condition deteriorated, and he visited our hospital. He had no abnormal findings on physical examination. A chest radiograph showed pneumonia in whole lung and CT showed multiple nodules and diffused ground glass opacities in both lung fields. Lung biopsy confirmed a diagnosis of Leydig cell tumor with lung metastasis. The diagnosis is based on the histopathology and immunohistochemistry.

Metastasized Leydig cell tumor in a dog.

We present the clinical findings, diagnosis and treatment of an 11-year old intact male Fox Terrier with a malignant Leydig cell tumor of the right testicle, which metastasized to the skeletal musculature of the left hind limb. The primary tumor and the metastasis were resected with narrow margins. The dog was treated with metronomic chemotherapy using thalidomid and dyclophosphamide. Local recurrence at the site of the metastasis and a pulmonary metastasis were present 30 months after surgery. The dog was euthanized.

Recurrent malignant Leydig cell tumor of testis: a case report with review of literature.

Malignant Testicular Leydig Cell tumors (leydigomas) are extremely rare to occur and mostly carry a bad prognosis. Here we describe the disease course of a middle aged patient with recurrent / metastatic Leydig cell tumor of testes, who needed repeated oncosurgical intervention and chemotherapy.

Spinal cord compression by a solitary metastasis from a low grade leydig cell tumour: a case report and review of the literature.

BACKGROUND: Leydig tumour is rare and there are only three cases with metastatic disease reported. CASE PRESENTATION: A 52 year-old Caucasian male was admitted, on emergency basis to the Orthopaedic Department with six weeks history of increasing midthoracic back pain, change in gait, poor balance, subjective weakness and numbness of the lower trunk and legs. MRI scan showed change in the signal intensity of T4 and T5 vertebral body but their height were maintained. Urgent T4 and T5 corpectomies, decompression of the spinal cord and reconstruction of the vertebral bodies were performed followed by radiotherapy. Neurological status significantly improved with a mild residual numbness over the dorsum of the right foot. The histology of the excised tumour was identical to the primary. At 2 years follow-up visit the patient is neurologically stable and disease free without other organs metastases. CONCLUSION: This is the first case in English literature, which shows that spinal metastases could occur even in the early stage of Leydig cell tumour, without other organs involvement. Aggressive surgical management of spinal metastases combined with post operative radiotherapy can give a better chance for long survivorship.

Fine needle aspiration of metastatic malignant Leydig cell tumor of testis: a case report.

A 53-year-old male presented with a right inguinal mass of one-year duration. The fine needle aspiration of the inguinal mass showed a highly cellular tumor composed of sheets and isolated, large round to polygonal cells with moderate pleomorphism. Many bare nuclei were seen with occasional intranuclear inclusions. A provisional diagnosis of metastasis probably of testicular tumour was made. The orchidectomy showed a brown tumor replacing the entire testis and infiltrating the epididymis. The histological features showed Leydig cell tumor without Reinke crystalloids.

Lack of efficacy of imatinib in a patient with metastatic Leydig cell tumor.

Imatinib is a tyrosine kinase inhibitor with activity in gastrointestinal stromal tumor and a variety of other solid and hematological malignancies. Studies in vitro and in a mouse model suggested that the imatinib might also be active in malignant Leydig cell tumor. We report on the--to our knowledge--first treatment experiment with imatinib in a patient with metastatic Leydig cell tumor. Unfortunately, the tumor progressed during treatment.

Non islet cell tumor hypoglycaemia in a metastatic Leydig cell tumor.

Non islet cell tumour hypoglycaemia (NICTH) is a rare cause of hypoglycaemia associated with malignancy and can be considered as a paraneoplastic syndrome. The hormonal factor associated with this condition is big IGF II, which exerts negative feedback effect and decreases the production of growth hormone and insulin. Due to low growth hormone levels, hepatic production of IGFBP 3 (the main binding protein of IGF II) is impaired. Excess free big IGF II is thus available for binding with insulin receptors to cause hypoglycaemia. Treatment options are either surgical removal of the tumour, administration of growth hormone, glucocorticoids or combination of treatments. A case of metastatic Leydig cell tumour causing NICTH has been discussed and the mechanism of NICTH hypoglycaemia and the treatment is outlined.

[Bilateral Leydig cell tumor of the test: a case report]. / Tumeur testiculaire bilatérale à cellules de Leydig: à propos d'un cas.

Testicular Leydig cell tumours are uncommon. Bilateral synchronous lesions are exceptional. They cause isosexual pseudo precocious puberty in childhood. The histological diagnosis of malignancy is sometimes difficult to establish and it can be made retrospectively when lymph nodes involvement or visceral metastasis appear in the follow-up. We report a case of a 9 year-old boy presenting bilateral Leydig cell tumour of the testis treated by bilateral radical orchiectomy who developed 2 years after the intervention a pulmonary metastasis.

Metastatic Leydig cell tumor of the testicle in a young African American male.

Malignant Leydig cell tumor (LCT) of the testis are extremely rare and account for less than 0.2% of all testicular cancers. Testicular tumors of all histological types rarely occur in African American men. The authors describe a rare case of an advanced stage malignant LCT arising from the testicle of an African American man at the young age of 35, who presented with hemoptysis and a productive cough. Clinical features and treatment of Leydig cell tumor of the testis are discussed.

Malignant Leydig cell tumour of the testis.

A case of malignant Leydig cell tumour is presented. It is a rare primary malignant tumour of the testis and occurs exclusively in adults. The present case is of interest because it occurred at the young age of 25 years which is rare. Histologically it showed almost all features which suggest malignancy and also had metastases to the lungs and liver. The clinical details and pathology of this tumour are discussed.

High frequency of metastatic Leydig cell testicular tumours.

690 patients were treated for testicular tumour in the course of 18 years. The histology of 7 cases showed Leydig cell tumour. In 4 of the 7 cases, a metastatic process leading to death was observed. At the time of diagnosis, 5 patients were found to have low stage, whereas 2 of the patients had advanced lymphatic involvement. The hematogenous and lymphatic metastases proved to be resistant to chemotherapy. Contrary to the major part of the literature, retroperitoneal lymphadenectomy should be performed with this histological type for the exact pathological staging immediately following orchiectomy.

Cushing syndrome as the presenting feature of metastatic Leydig cell tumor of the testis.

We report a patient with a history of orchiectomy for Leydig cell tumor of the testis who developed Cushing syndrome. This syndrome was due to ectopic production of cortisol and was the primary feature of tumor recurrence.

Metastatic Leydig cell tumor with sarcomatoid differentiation.

Leydig cell tumors of the testis are uncommon. Only about 10% of cases have a malignant course. It has been stated that the only definite criterion for malignancy is presence of metastasis. We present a 47-year-old patient with metastatic Leydig cell tumor 17 years after initial diagnosis, to our knowledge the longest reported interval between diagnosis and the development of metastasis. The primary tumor did not exhibit convincing features of malignancy. The initial metastasis in the right perirenal fat tissue showed a biphasic tumor with sarcomatoid differentiation not described previously in a metastatic Leydig cell tumor.

[Malignant Leydig cell tumor. Report of a case and review of the literature]. / Tumor maligno de células de Leydig. Presentación de un caso y revisión de la literatura.

OBJECTIVE: To report an additional case of malignant Leydig cell tumor of the testis. The clinical, hormonal and histological criteria for malignancy are analyzed and the different therapeutic options are discussed. METHODS/RESULTS: A 75-year-old male presented with a painful testicular tumor, dyspnea and generalized syndrome. The chest film showed multiple bilateral metastatic pulmonary lesions. Pathological analysis disclosed malignant Leydig cell tumor. The patient died one month after orchidectomy due to the neoplasm. CONCLUSIONS: Metastatic Leydig cell tumor is uncommon. To date, no available treatment can significantly alter the course of the disease.