CASE REPORT
Rectal Lymphogranuloma Venereum in HIV-infected Patients Can Mimic Lymphoma Etienne Crickx, MD,* Ve´ronique Meignin, MD,w Laurence Ge´rard, MD,* Isabelle Plantier-Colcher, MD,z Francine Walker-Combrouze, MD,y David Boutboul, MD,* Lionel Galicier, MD,* Claire Fieschi, MD, PhD,* and Eric Oksenhendler, MD, PhD*
Abstract: An outbreak of rectal lymphogranuloma venereum (LGV) has been reported since 2003 in men who have sex with men, most of them being infected with human immunodeficiency virus. In these patients, unusual clinical presentations such as rectal tumor or intense lymphoproliferation on rectal biopsies may lead to an erroneous diagnosis of aggressive non-Hodgkin lymphoma. Three patients were referred to our center for the management of rectal B-cell non-Hodgkin lymphoma on the basis of a rectal pathologic specimen showing intense lymphoproliferation, the very suspect of lymphoma. Because of anamnesis of anal intercourses and venereal diseases, additional study revealed that all 3 had a positive Chlamydia trachomatis polymerase chain reaction on the rectal biopsy specimen. Rectal LGV was therefore considered and successfully treated with antibiotics. We propose that all patients presenting with a suspected rectal lymphoma should have a careful anamnesis of sexual behavior and a specific detection of C. trachomatis using polymerase chain reaction analysis on biopsy specimen to rule out the possibility of rectal LGV. Key Words: Chlamydia trachomatis, lymphogranuloma venereum, lymphoma, human immunodeficiency virus
(J Clin Gastroenterol 2016;50:414–416)
L
ymphogranuloma venereum (LGV) is a venereal disease caused by Chlamydia trachomatis serovars L1, L2, or L3.1 The infection begins with genital ulcer, followed a few weeks later by inguinal lymphadenopathies. Since 2003, an outbreak of rectal LGV caused by L2b serovar has been reported in Europe and Northern America in men who have sex with men, most of them being infected with human immunodeficiency virus (HIV).2–4 Clinical symptoms are those of acute proctitis, that is, pain, rectal bleeding, or diarrhea.3 Histopathologic findings include inflammatory infiltrate with microabcesses, epithelioid granuloma, and necrosis.5 In all cases, cure can be achieved with a 2 to 3 weeks course of doxycycline.6 HIV-infected patients have an increased risk to develop Hodgkin and non-Hodgkin From the *Department of Clinical Immunology, Service d’Immunopathologie Clinique; wDepartment of Pathology, Hoˆpital Saint-Louis; yDepartment of Pathology, Hoˆpital Bichat, AP-HP, Universite´ Paris7 Diderot, Paris; and zDepartment of Clinical Hematology, Hoˆpital Victor Provo, Roubaix, France. E.C. wrote the manuscript and realized data acquisition. V.M., L.G., I.P.-C., F.W.-C., D.B., L.G., C.F., and E.O. made critical revision of the manuscript. The authors declare that they have nothing to disclose. Reprints: Etienne Crickx, MD, Department of Clinical Immunology, Service d’Immunopathologie Clinique, Hoˆpital Saint-Louis, 1 Avenue Claude Vellefaux, Paris 75475 Cedex 10, France (e-mail: [email protected]). Copyright r 2015 Wolters Kluwer Health, Inc. All rights reserved.
lymphomas. In anorectal localization, one of the subtype of lymphoma encountered is plasmablastic lymphoma, characterized by an aggressive proliferation of plasmablastic cells with positivity of CD138 staining and Epstein-Barr virus (EBV)encoded small RNAs (EBER) in situ hybridization.7,8 Three patients were referred to our center for the management of rectal B-cell non-Hodgkin lymphoma on the basis of a rectal pathologic specimen showing intense lymphoproliferation, the very suspect of lymphoma. Additional study revealed that all 3 had a positive C. trachomatis polymerase chain reaction (PCR) on the rectal biopsy specimen. Rectal LGV was therefore considered and successfully treated with antibiotics.
CASE REPORTS Case 1 A 43-year-old man presented with rectal bleeding and pruritus ani. He was known for HIV infection and was treated with combined antiretroviral therapy. The CD4 + cell count was 484106 cells/L and the plasma HIV-RNA level was
Rectal Lymphogranuloma Venereum in HIV-infected Patients Can Mimic Lymphoma Etienne Crickx, MD,* Ve´ronique Meignin, MD,w Laurence Ge´rard, MD,* Isabelle Plantier-Colcher, MD,z Francine Walker-Combrouze, MD,y David Boutboul, MD,* Lionel Galicier, MD,* Claire Fieschi, MD, PhD,* and Eric Oksenhendler, MD, PhD*
Abstract: An outbreak of rectal lymphogranuloma venereum (LGV) has been reported since 2003 in men who have sex with men, most of them being infected with human immunodeficiency virus. In these patients, unusual clinical presentations such as rectal tumor or intense lymphoproliferation on rectal biopsies may lead to an erroneous diagnosis of aggressive non-Hodgkin lymphoma. Three patients were referred to our center for the management of rectal B-cell non-Hodgkin lymphoma on the basis of a rectal pathologic specimen showing intense lymphoproliferation, the very suspect of lymphoma. Because of anamnesis of anal intercourses and venereal diseases, additional study revealed that all 3 had a positive Chlamydia trachomatis polymerase chain reaction on the rectal biopsy specimen. Rectal LGV was therefore considered and successfully treated with antibiotics. We propose that all patients presenting with a suspected rectal lymphoma should have a careful anamnesis of sexual behavior and a specific detection of C. trachomatis using polymerase chain reaction analysis on biopsy specimen to rule out the possibility of rectal LGV. Key Words: Chlamydia trachomatis, lymphogranuloma venereum, lymphoma, human immunodeficiency virus
(J Clin Gastroenterol 2016;50:414–416)
L
ymphogranuloma venereum (LGV) is a venereal disease caused by Chlamydia trachomatis serovars L1, L2, or L3.1 The infection begins with genital ulcer, followed a few weeks later by inguinal lymphadenopathies. Since 2003, an outbreak of rectal LGV caused by L2b serovar has been reported in Europe and Northern America in men who have sex with men, most of them being infected with human immunodeficiency virus (HIV).2–4 Clinical symptoms are those of acute proctitis, that is, pain, rectal bleeding, or diarrhea.3 Histopathologic findings include inflammatory infiltrate with microabcesses, epithelioid granuloma, and necrosis.5 In all cases, cure can be achieved with a 2 to 3 weeks course of doxycycline.6 HIV-infected patients have an increased risk to develop Hodgkin and non-Hodgkin From the *Department of Clinical Immunology, Service d’Immunopathologie Clinique; wDepartment of Pathology, Hoˆpital Saint-Louis; yDepartment of Pathology, Hoˆpital Bichat, AP-HP, Universite´ Paris7 Diderot, Paris; and zDepartment of Clinical Hematology, Hoˆpital Victor Provo, Roubaix, France. E.C. wrote the manuscript and realized data acquisition. V.M., L.G., I.P.-C., F.W.-C., D.B., L.G., C.F., and E.O. made critical revision of the manuscript. The authors declare that they have nothing to disclose. Reprints: Etienne Crickx, MD, Department of Clinical Immunology, Service d’Immunopathologie Clinique, Hoˆpital Saint-Louis, 1 Avenue Claude Vellefaux, Paris 75475 Cedex 10, France (e-mail: [email protected]). Copyright r 2015 Wolters Kluwer Health, Inc. All rights reserved.
lymphomas. In anorectal localization, one of the subtype of lymphoma encountered is plasmablastic lymphoma, characterized by an aggressive proliferation of plasmablastic cells with positivity of CD138 staining and Epstein-Barr virus (EBV)encoded small RNAs (EBER) in situ hybridization.7,8 Three patients were referred to our center for the management of rectal B-cell non-Hodgkin lymphoma on the basis of a rectal pathologic specimen showing intense lymphoproliferation, the very suspect of lymphoma. Additional study revealed that all 3 had a positive C. trachomatis polymerase chain reaction (PCR) on the rectal biopsy specimen. Rectal LGV was therefore considered and successfully treated with antibiotics.
CASE REPORTS Case 1 A 43-year-old man presented with rectal bleeding and pruritus ani. He was known for HIV infection and was treated with combined antiretroviral therapy. The CD4 + cell count was 484106 cells/L and the plasma HIV-RNA level was
