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Hamartoma of the Epididymis Simulating Tuberculous Epididymitis
THE JOURNAL OF UROLOGY
Vol. 91, No. 5 May 1964 Copyright © 1964 by The Williams & Wilkins Co. Printed in U.S.A.
HAMARTOMA OF THE EPIDIDYMIS SIMULATING TUBERCULOUS EPIDIDYMITIS DANIEL JICHLINSKI
AND
NEIL WARD-McQUAID
From the Mansfield Group of Hospitals, Nottinghamshire, England
Tumors of the epididymis are uncommon; less than 100 cases have been reported. Albrecht applied the term hamartoma to certain tumorlike dysplasias characterized by a quantitative increase in the tissue normally present at a particular site. 1 These hamartomas differ from true neoplasms in that there are no qualitative alterations except those due to the growth of the malformation.
Fm. 1. Cut surface of hemocapillary hamartoma of epididymis. CASE REPORT
A 28-year-old insurance agent complained of a dragging feeling and swelling of his left scrotum a few weeks in duration. He had no other symptoms. At the age of 2 months, the patient had had a lymphangioma excised from the left side of his chest wall by the late Mr. Alex McLennan at the Royal Hospital for Sick Children, Glasgow. This lymphangioma extended into the axilla and ramified through the pectoral and latissismus dorsi muscles. Seven years later a recurrence of the mass was treated by deep radiotherapy. Gigantism of the first and second toes of his left foot with increased length of the leg was reAccepted for publication December 16, 1963. 1 Albrecht: Ueber Hamartome. Verhandl. dtsch. ges. Path., 7: 153-157, 1904.
corded at this time and these toes were later excised. He then noticed a further small lesion on the left side of the abdomen resembling that previously treated on his chest. When the patient was 26 years old a chronic cough developed. The patient then underwent tests including a normal erythrocyte sedimentation rate, chest x-ray and sputum examination for pulmonary tuberculosis, with negative results. The Heaf test, however, was positive. There was no family history of tuberculosis. The patient, on examination, appeared to be a fit young man. The only abnormalities were the scar of the previous operation on his chest and the presumably lymphangiomatous lesion on the left side of the abdomen, together with a firm enlargement of the whole of the left epididymis, except the globus minor. The prostate, vesicles, testes and vasa were normal, but the left cord was thickened. Urine deposit and culture were negative, including examination for tubercle bacilli. The vVassermann reaction test was also negative. The likelihood of another tumor similar to that on the abdomen and to that already excised from the chest wall was considered. It was thought wise to make a provisional diagnosis of tuberculosis of the epididymis and to give a course of antituberculous drugs (streptomycin, isoniazid, and para-aminosalicylic acid) in the usual dosage. After some weeks there was no change in the epididymis and operation was then advised. With the patient under general anesthesia the left testicle was exposed. The testis and globus minor were normal. There was enlargement of the globus major and most of the epididymis with extension into the distal part of the cord. The condition resembled a hemolymphangio-lipomatous formation. Epididymectomy was performed. Convalescence was uneventful. The excised epididymis measured 7.5 by 4 cm. (fig. 1). Dr. A. B. Hill reported the microscopic findings (fig. 2) as an epididymal enlargement due to the presence of a hemocapillary hamartoma.
576
HAMARTOMA OF EPIDIDYMIS
577
Fm. 2. Low power microscopic appearances of hemocapillary hamartoma of epididymis DISCUSSION
Review of the literature reveals no agreement on either the origin or nomenclature of benign tumors of the epididymis. More recently Catania 2 and others have distinguished hamartomas from true tumors. Previously benign tumors were classified morphologically, distinguishing for example the fibrolipoma from the lymphangioma, the hemangioma and so on, or according to their supposed origin, using terms such as mesothelioma. Such tumors have also been referred to as angiomatoid formations, 3 or adenomatoid tumors of the epididymis. 4 Inflammatory tumors and granulomas, and other such epididymal swellings must also be distinguished from both the hamartomas and the true epididymal tumors and are not discussed further. Endothelial, mesothelial and epithelial origins 2 Catania, V.: Racemose angioma of the epididymis. Arch. ital. Ural., 28: 187-192, 1955. 3 Morehead, R. P.: Angromatoid formations in the genital organs with and without tumor formation. Arch. Path., 42: 56-63, 1946. 4 Golden, A. and Ash, J. E.: Adenomatoid tumors of genital tract. Amer. J. Path., 21: 63-79, 1945.
have all been suggested for what are now generally called epididymal hamartomas. Naegeli 5 was the first of several authors, including Longo and associates 6 to suggest the mesonephric system as the source of these tumors. Sundarasivarao7 and Willis8 produced very convincing evidence that these dysplasias arose from miillerian vestiges. None of the 7 vascular tumors of the epididymis described in the literature as hemangiomas by Hardouin, 9 Hosford, 10 cl' Abreu, 11 Thompson, 12 5 Naegeli, T.: Ein Mischtumor des Sarnenstranges. Virchow Arch., 208: 364-367, 1912. 6 Longo, V. J., McDonald, J. R. and Thompson, G. J.: Primary neoplasms of the epididymis; special reference to adenomatoid tumors. J. A. M. A., 147: 937-941, 1951. 7 Sundarasivarao, D.: The mullerian vestiges and benign epithelial tumors of the epididymis. J. Path. & Bact., 66: 417-432, 1953. 8 Willis, R. A.: Pathology of Tumours, 2nd ed. London: Butterworth & Co., 1953. 9 Hardouin: Resection sub to tale de la diaphyse tibiale dans un cas d'osteomye'lite aigue. Resultar au bout de sept aus. Bull. Soc. Chir. Paris: 37: 1358-1363, 1911. 10 Hosford, J. P.: Angioma of epididymis. Proc. Roy. Soc. Med., 25: 539, 1931-32.
578
JICHLINSKI AND WARD-MCQUAID
(1936) and as a hamartoma by Catania, 2 showed the hemocapillary structure of this case, nor were they found in a patient suffering from hamartomas.
11 d'Abreu, A. L.: Haemangioma of the epididymis. Brit. J. Surg., 23: 858-859, 1936. 12 Thompson, G. J.: Tumors of the spermatic cord, epididymis and testicular tunics; a review of literature and report of forty-one cases. Surg., Gynec. & Obst., 62: 712-728, 1936.
SUMMARY
In the patient reported a hemocapillary hamartoma of the epididymis simulated tuberculous epididymitis. There had been a history of a previous lymphangiomatous hamartoma of the chest wall and abdomen, and also a chronic cough. We are indebted to Dr. A. B. Hill for the pathology report, and the Royal Hospital for Sick Children, Glasgow for details from its records.
Vol. 91, No. 5 May 1964 Copyright © 1964 by The Williams & Wilkins Co. Printed in U.S.A.
HAMARTOMA OF THE EPIDIDYMIS SIMULATING TUBERCULOUS EPIDIDYMITIS DANIEL JICHLINSKI
AND
NEIL WARD-McQUAID
From the Mansfield Group of Hospitals, Nottinghamshire, England
Tumors of the epididymis are uncommon; less than 100 cases have been reported. Albrecht applied the term hamartoma to certain tumorlike dysplasias characterized by a quantitative increase in the tissue normally present at a particular site. 1 These hamartomas differ from true neoplasms in that there are no qualitative alterations except those due to the growth of the malformation.
Fm. 1. Cut surface of hemocapillary hamartoma of epididymis. CASE REPORT
A 28-year-old insurance agent complained of a dragging feeling and swelling of his left scrotum a few weeks in duration. He had no other symptoms. At the age of 2 months, the patient had had a lymphangioma excised from the left side of his chest wall by the late Mr. Alex McLennan at the Royal Hospital for Sick Children, Glasgow. This lymphangioma extended into the axilla and ramified through the pectoral and latissismus dorsi muscles. Seven years later a recurrence of the mass was treated by deep radiotherapy. Gigantism of the first and second toes of his left foot with increased length of the leg was reAccepted for publication December 16, 1963. 1 Albrecht: Ueber Hamartome. Verhandl. dtsch. ges. Path., 7: 153-157, 1904.
corded at this time and these toes were later excised. He then noticed a further small lesion on the left side of the abdomen resembling that previously treated on his chest. When the patient was 26 years old a chronic cough developed. The patient then underwent tests including a normal erythrocyte sedimentation rate, chest x-ray and sputum examination for pulmonary tuberculosis, with negative results. The Heaf test, however, was positive. There was no family history of tuberculosis. The patient, on examination, appeared to be a fit young man. The only abnormalities were the scar of the previous operation on his chest and the presumably lymphangiomatous lesion on the left side of the abdomen, together with a firm enlargement of the whole of the left epididymis, except the globus minor. The prostate, vesicles, testes and vasa were normal, but the left cord was thickened. Urine deposit and culture were negative, including examination for tubercle bacilli. The vVassermann reaction test was also negative. The likelihood of another tumor similar to that on the abdomen and to that already excised from the chest wall was considered. It was thought wise to make a provisional diagnosis of tuberculosis of the epididymis and to give a course of antituberculous drugs (streptomycin, isoniazid, and para-aminosalicylic acid) in the usual dosage. After some weeks there was no change in the epididymis and operation was then advised. With the patient under general anesthesia the left testicle was exposed. The testis and globus minor were normal. There was enlargement of the globus major and most of the epididymis with extension into the distal part of the cord. The condition resembled a hemolymphangio-lipomatous formation. Epididymectomy was performed. Convalescence was uneventful. The excised epididymis measured 7.5 by 4 cm. (fig. 1). Dr. A. B. Hill reported the microscopic findings (fig. 2) as an epididymal enlargement due to the presence of a hemocapillary hamartoma.
576
HAMARTOMA OF EPIDIDYMIS
577
Fm. 2. Low power microscopic appearances of hemocapillary hamartoma of epididymis DISCUSSION
Review of the literature reveals no agreement on either the origin or nomenclature of benign tumors of the epididymis. More recently Catania 2 and others have distinguished hamartomas from true tumors. Previously benign tumors were classified morphologically, distinguishing for example the fibrolipoma from the lymphangioma, the hemangioma and so on, or according to their supposed origin, using terms such as mesothelioma. Such tumors have also been referred to as angiomatoid formations, 3 or adenomatoid tumors of the epididymis. 4 Inflammatory tumors and granulomas, and other such epididymal swellings must also be distinguished from both the hamartomas and the true epididymal tumors and are not discussed further. Endothelial, mesothelial and epithelial origins 2 Catania, V.: Racemose angioma of the epididymis. Arch. ital. Ural., 28: 187-192, 1955. 3 Morehead, R. P.: Angromatoid formations in the genital organs with and without tumor formation. Arch. Path., 42: 56-63, 1946. 4 Golden, A. and Ash, J. E.: Adenomatoid tumors of genital tract. Amer. J. Path., 21: 63-79, 1945.
have all been suggested for what are now generally called epididymal hamartomas. Naegeli 5 was the first of several authors, including Longo and associates 6 to suggest the mesonephric system as the source of these tumors. Sundarasivarao7 and Willis8 produced very convincing evidence that these dysplasias arose from miillerian vestiges. None of the 7 vascular tumors of the epididymis described in the literature as hemangiomas by Hardouin, 9 Hosford, 10 cl' Abreu, 11 Thompson, 12 5 Naegeli, T.: Ein Mischtumor des Sarnenstranges. Virchow Arch., 208: 364-367, 1912. 6 Longo, V. J., McDonald, J. R. and Thompson, G. J.: Primary neoplasms of the epididymis; special reference to adenomatoid tumors. J. A. M. A., 147: 937-941, 1951. 7 Sundarasivarao, D.: The mullerian vestiges and benign epithelial tumors of the epididymis. J. Path. & Bact., 66: 417-432, 1953. 8 Willis, R. A.: Pathology of Tumours, 2nd ed. London: Butterworth & Co., 1953. 9 Hardouin: Resection sub to tale de la diaphyse tibiale dans un cas d'osteomye'lite aigue. Resultar au bout de sept aus. Bull. Soc. Chir. Paris: 37: 1358-1363, 1911. 10 Hosford, J. P.: Angioma of epididymis. Proc. Roy. Soc. Med., 25: 539, 1931-32.
578
JICHLINSKI AND WARD-MCQUAID
(1936) and as a hamartoma by Catania, 2 showed the hemocapillary structure of this case, nor were they found in a patient suffering from hamartomas.
11 d'Abreu, A. L.: Haemangioma of the epididymis. Brit. J. Surg., 23: 858-859, 1936. 12 Thompson, G. J.: Tumors of the spermatic cord, epididymis and testicular tunics; a review of literature and report of forty-one cases. Surg., Gynec. & Obst., 62: 712-728, 1936.
SUMMARY
In the patient reported a hemocapillary hamartoma of the epididymis simulated tuberculous epididymitis. There had been a history of a previous lymphangiomatous hamartoma of the chest wall and abdomen, and also a chronic cough. We are indebted to Dr. A. B. Hill for the pathology report, and the Royal Hospital for Sick Children, Glasgow for details from its records.