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Cytomegalovirus and lymphoma in a pediatric transplant recipient
494
Letters to the Editor
DISCUSSION XX males have male pheaotype, male psychosexual identification, gonads of testicular type without evidence of ovarian tissue, and XX sex chromosomes. They have no female sex structures. Clinically they closely resemble individuals with Klinefelter syndrome. Although we were unable to find our patient's gonads, his response to chorionic gonadotropin indicated the presence of occult testicular tissue; there was no indication of occult ovarian tissue. Since he fulfills all other criteria for the diagnosis, we believe, he is most likely an XX male. The chemical tests, however, do not prove the absence of any ovarian tissue, so we cannot completely exclude the diagnosis of a true hermaphrodite. George M. Lazarus, M.D. Major, MC, USAF Department of Pediatrics Robert D. Rodner, M.D. Major, MC, USA F Department of Urology USA F Medical Center Wright-Patterson Air Force Base Ohio, 45433 REFERENCES
1. de la Chapelle A: Nature and origin of males with XX sex chromosomes, Am J Hum Genet 24:71, 1972. 2. Levine L, and New M: Preoperative detection of hidden testes, Am J Dis Child 121:176, 1971.
Cytomegalovirus and lymphoma in a pediatric transplant recipient To the Editor: We have recently observed an 11-year-old girl who presented with disseminated cytomegalovirus infection one year posttrans-
The Journal of Pediatrics September 1975
plant and at autopsy was found to have an unsuspected lympho. proliferative neoplasm, raising the question of whether the pathogenesis of this tumor was related to the viral infection or to an abnormal response to the viral infection. CASE REPORT
A 10-year-old girl with renal failure secondary to hypocomple. mentemic membranoproliferative glomerulonephritis received a kidney transplant from her mother. In the first posttransplant year she had two rejection episodes which responded rapidly to treatment. Routine allograft biopsy performed one year following transplantation revealed changes consistent with mild chronic rejection. Thirteen months following transplantation she developed weakness, lethargy, urticarial rash, and spiking temperatures to 104~ F. Physical examination was normal except for bilateral rhles and rhonchi. A roentgenogram of the chest (Fig. 1) showed a bilateral nodular infiltrate, and at bronchoscopy a creamy white exudate was seen in both mainstem bronchi. Open lung biopsy was performed to define a presumptive opportunistic infection, but histologic sections were reported to show only a histiocytic infiltrate consistent with chronic inflammation. She had a progressive downhill course and developed leukopenia, anorexia, and a complete heart block with an electrocardiogram consistent with viral myocardifis. Cultures of urine, sputum, bronchial washings, and lung biopsy grew cytomegalovirus. Fifty days following admission she had a Jacksonian seizure with an associated cardiorespiratory arrest from which she could not be resuscitated and died on the fifty-ninth hospital day. Autopsy revealed cytomegalovirus infection of lungs, liver, heart, and esophagus. An unexpected finding was a widely disseminated malignant lymphoma, "histiocytie" in type? There was involvement of brain, lung, fiver, kidney, myocardium, and lymph nodes. COMMENT An increased incidence of cancer has been reported in transplant recipients. 2 Lymphoproliferative tumors and carcinoma of the skin and cervix comprise 75% of these tumors. In addition transplant recipients have a high incidence (up to 90%) of herpesvirus infections.~ Herpesviruses have been implicated in
Fig. 1. Roentgenogram of the chest taken on admission to hospital, 13 months after transplantation, showing bilateral nodular infiltrates. These were shown at autopsy to be foci of lymphoma.
Volume 87 Number 3
the etiology of both lymphoproliferative tumors and carcinoma of the skin and cervix in both animals and man. 4-6 We report a case of concurrent lymphoma and herpesvirus infection. Is it possible that herpesvirus infection plays a role in the increased incidence of malignancy in the transplant recipient? Arthur J. Matas, M.D. Surgical Resident Richard L. Simmons, M.D. Professor of Surgery and Microbiology John H. Kersey, M.D. Associate Professor of Lab Medicine, Pathology and Pediatrics Carl M. Kjellstrand, M.D. Professor of Medicine and Surgery John S. Najarian, M.D. Professor and Chairman of Surgery Mayo Memorial Bldg. University of Minnesota Minneapolis, Minn. 55455
Letters to the Editor
495
REFERENCES
1. Rappaport H: Malignant lymphomas, in Atlas of pathology, Section III, Fascicle 8, Tumors of the hematopoietic system, Washington, D.C. 1966, Armed Forces Institute of Pathology, p 61. 2. Penn I, and Starzl TE: Malignant tumors arising de novo in immunosuppressed organ transplant recipients, Transplantation 14:407, 1972. 3. Simmons RL, Lopez C, Balfour H, Jr, Kalis J, Rattazzi LC, and Najarian JS: Cytomegalovirus: Clinical virological correlations in renal transplant recipients, Ann Surg 180:623, 1974. 4. Klein G: Herpesviruses and oncogenesis, Proc Nat Acad Sci 69:1056, 1972. 5. Rapp F: Question: Do herpesviruses cause cancer? Answer: Of course they do! J Natl Ca Inst 50:825, 1973. 6. Wyburn-Mason R: Malignant change following herpes simplex, Br Med J 2:615, 1957.
Letters to the Editor
DISCUSSION XX males have male pheaotype, male psychosexual identification, gonads of testicular type without evidence of ovarian tissue, and XX sex chromosomes. They have no female sex structures. Clinically they closely resemble individuals with Klinefelter syndrome. Although we were unable to find our patient's gonads, his response to chorionic gonadotropin indicated the presence of occult testicular tissue; there was no indication of occult ovarian tissue. Since he fulfills all other criteria for the diagnosis, we believe, he is most likely an XX male. The chemical tests, however, do not prove the absence of any ovarian tissue, so we cannot completely exclude the diagnosis of a true hermaphrodite. George M. Lazarus, M.D. Major, MC, USAF Department of Pediatrics Robert D. Rodner, M.D. Major, MC, USA F Department of Urology USA F Medical Center Wright-Patterson Air Force Base Ohio, 45433 REFERENCES
1. de la Chapelle A: Nature and origin of males with XX sex chromosomes, Am J Hum Genet 24:71, 1972. 2. Levine L, and New M: Preoperative detection of hidden testes, Am J Dis Child 121:176, 1971.
Cytomegalovirus and lymphoma in a pediatric transplant recipient To the Editor: We have recently observed an 11-year-old girl who presented with disseminated cytomegalovirus infection one year posttrans-
The Journal of Pediatrics September 1975
plant and at autopsy was found to have an unsuspected lympho. proliferative neoplasm, raising the question of whether the pathogenesis of this tumor was related to the viral infection or to an abnormal response to the viral infection. CASE REPORT
A 10-year-old girl with renal failure secondary to hypocomple. mentemic membranoproliferative glomerulonephritis received a kidney transplant from her mother. In the first posttransplant year she had two rejection episodes which responded rapidly to treatment. Routine allograft biopsy performed one year following transplantation revealed changes consistent with mild chronic rejection. Thirteen months following transplantation she developed weakness, lethargy, urticarial rash, and spiking temperatures to 104~ F. Physical examination was normal except for bilateral rhles and rhonchi. A roentgenogram of the chest (Fig. 1) showed a bilateral nodular infiltrate, and at bronchoscopy a creamy white exudate was seen in both mainstem bronchi. Open lung biopsy was performed to define a presumptive opportunistic infection, but histologic sections were reported to show only a histiocytic infiltrate consistent with chronic inflammation. She had a progressive downhill course and developed leukopenia, anorexia, and a complete heart block with an electrocardiogram consistent with viral myocardifis. Cultures of urine, sputum, bronchial washings, and lung biopsy grew cytomegalovirus. Fifty days following admission she had a Jacksonian seizure with an associated cardiorespiratory arrest from which she could not be resuscitated and died on the fifty-ninth hospital day. Autopsy revealed cytomegalovirus infection of lungs, liver, heart, and esophagus. An unexpected finding was a widely disseminated malignant lymphoma, "histiocytie" in type? There was involvement of brain, lung, fiver, kidney, myocardium, and lymph nodes. COMMENT An increased incidence of cancer has been reported in transplant recipients. 2 Lymphoproliferative tumors and carcinoma of the skin and cervix comprise 75% of these tumors. In addition transplant recipients have a high incidence (up to 90%) of herpesvirus infections.~ Herpesviruses have been implicated in
Fig. 1. Roentgenogram of the chest taken on admission to hospital, 13 months after transplantation, showing bilateral nodular infiltrates. These were shown at autopsy to be foci of lymphoma.
Volume 87 Number 3
the etiology of both lymphoproliferative tumors and carcinoma of the skin and cervix in both animals and man. 4-6 We report a case of concurrent lymphoma and herpesvirus infection. Is it possible that herpesvirus infection plays a role in the increased incidence of malignancy in the transplant recipient? Arthur J. Matas, M.D. Surgical Resident Richard L. Simmons, M.D. Professor of Surgery and Microbiology John H. Kersey, M.D. Associate Professor of Lab Medicine, Pathology and Pediatrics Carl M. Kjellstrand, M.D. Professor of Medicine and Surgery John S. Najarian, M.D. Professor and Chairman of Surgery Mayo Memorial Bldg. University of Minnesota Minneapolis, Minn. 55455
Letters to the Editor
495
REFERENCES
1. Rappaport H: Malignant lymphomas, in Atlas of pathology, Section III, Fascicle 8, Tumors of the hematopoietic system, Washington, D.C. 1966, Armed Forces Institute of Pathology, p 61. 2. Penn I, and Starzl TE: Malignant tumors arising de novo in immunosuppressed organ transplant recipients, Transplantation 14:407, 1972. 3. Simmons RL, Lopez C, Balfour H, Jr, Kalis J, Rattazzi LC, and Najarian JS: Cytomegalovirus: Clinical virological correlations in renal transplant recipients, Ann Surg 180:623, 1974. 4. Klein G: Herpesviruses and oncogenesis, Proc Nat Acad Sci 69:1056, 1972. 5. Rapp F: Question: Do herpesviruses cause cancer? Answer: Of course they do! J Natl Ca Inst 50:825, 1973. 6. Wyburn-Mason R: Malignant change following herpes simplex, Br Med J 2:615, 1957.