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Castleman disease in pregnancy
10. Beguin Y, Loo M, R’Zik S, Sautois B, Lejeune F, Rorive G, et al. Quantitative assessment of erythropoiesis in haemodialysis patients demonstrates gradual expansion of erythroblasts during constant treatment with recombinant human erythropoietin. Br J Haematol 1995;89:17-23. 11. Garcia JF, Sherwood J, Golwasser E. Radioimmunoassay of erythropoietin. Blood Cells 1979;5:405-11. 12. Breymann C, Major A, Richter C, Huch R, Huch A. Recombinant human erythropoietin and parenteral iron in the treatment of pregnancy anemia: A pilot study. J Perinat Med 1995;23:88-98. 13. Shaefer RM, Hijrl WH, Massry SG. Treatment of renal anemia with recombinant human erythropoietin. Am J Nephrol 1989;9:353-62. 14. Harris SA, Payne G Jr, Putman JM. Erythropoietin treatment of erythropoietin-deficient anemia without renal disease during pregnancy. Obstet Gynecol 1996;87:812-4.
Address
CASTLEMAN
disease presenting as an abdominal bleeding during the second trimester
DISEASE
reprint
requests
to:
Nicola Carretti, MD Obstetrics and Gynecology Clinica Ostetrica e Ginecologica dell’Universitri Via Paolo Mascagni, 46 I-53100 Siena ltaly
di Siena
Received December 30, 1996. Received in revised form April 14, 1997. Accepted April 21,1997. Copyright 0 1997 by The American College of Obstetricians Gynecologists. Published by Elsevier Science Inc.
and
mass and vaginal of pregnancy.
IN PREGNANCY Yoram Abmmov, MD, Michel Nadjari, MD, Ayula Abrahamov, MD, Constantin Aeinus, MD, and Shaoul 0. Anteby, MD Background: Castleman disease, a rare disorder characterized by benign proliferation of lymphoid tissues, generally presents as a solitary mediastinal mass. We report the first case of this disease during pregnancy. Case: A 32-year-old woman presented with a large abdominal mass and vaginal bleeding during the second trimester of pregnancy. Abdominal ultrasound demonstrated a large, retroperitoneal solid mass of mixed echogenicity and increased vascularity. The patient underwent explorative laparotomy that revealed a mesenteric mass, histologically consistent with Castleman disease of the hyaline-vascular type. The mass was excised completely, and the immediate postoperative course was uneventful, although the patient went into spontaneous preterm labor during the 29th week of pregnancy. Conclusion: Castleman disease should be considered one of the benign etiologies for an abdominal or retroperitoneal mass during pregnancy. (Obstet Gynecol 1997;90:653-4. 0 1997 by The American College of Obstetricians and Gynecologists.) Castleman disease, a rare disorder characterized by benign proliferation of lymphoid tissue,’ usually presents as a localized soft-tissue mass in the neck or mediastinum. We report a case of mesenteric Castleman From fhe Departments of Obstetrics and Gynecology, and Pathology, Hadassah Hebrew University Medical Center, Ein-Kerem, and jrom the Department of Pediatrics, Shaare-Zedek Medical Center, ]erusalem, Israel.
VOL.9O,NO.4,PART
2,OCTOBER
1997
Case A 32-year-old eight, para
married, six, presented
generally healthy woman, gravida with vaginal bleeding during her
15th gestational week. Physical examination revealed a large, semisolid, partially fixed, nontender, irregularly outlined mass above and right of the uterus. There was a small amount of uterine bleeding, but the cervix was closed. Abdominal sonography indicated a single, live fetus consistent with the stage of gestation, with an adequate amount of amniotic fluid and no signs of placental separation. A large solid mass of mixed echogenicity and increased vascularity by color Doppler, 10 X 8 cm in diameter, was demonstrated in the right retroperitoneum, near the lower pole of the right kidney. The patient was hospitalized, and 10 days after vaginal bleeding subsided, she underwent an exploratory laparotomy. There
was
a 12-X-6-cm
solid
mass
near
the
root
of the
transverse mesocolon, adjacent to the superior mesenteric vessels and surrounded by loose connective tissue. No enlarged lymph nodes were palpated, and the genital and abdominal organs were normal. The mass was excised completely. A frozen section suggested a high probability of lymphoma, but the permanent histopathologic sections revealed numerous hyperplastic lymphoid follicles in a structure
of a lymph
node
covered
by a thin
capsule.
Germinal
centers were transfixed by penetrating vessels with foci of hyalinization, whereas mantle zones showed concentric hyperplasia (Figure 1). Interfollicular stroma contained abundant vessels, mature small lymphocytes, and almost no plasma cells. These findings are diagnostic for Castleman disease of the hyaline-vascular type. The patient had an uneventful postoperative course, although
12 weeks
later
she developed
uterine
contractions
and
delivered prematurely during the 29th week of pregnancy. The infant, weighing 1410 g at delivery, was admitted to the neonatal intensive care unit, where he did well and was discharged in good general condition 6 weeks later.
0029-7844/97/$17.00 PII SO'J29-7844(97)00222-6
653
Figure 1. Photomicrograph (X100) showing a lymph follicle composed of concentrically arranged small lymphocytes with a hyalinized germinal center penetrated by a number of small vessels. The interfollicular space is highly vascular and contains almost no plasma cells. These features are characteristic of Castleman disease of the hyalinevascular type.
Cowmen t Castleman disease, first described in 1956 after analysis of a number of cases of enlarged mediastinal lymph nodes that initially had been classified erroneously as thymic tumors,’ is attributed to lymph node hyperplasia of unknown etiology. The disorder is seen most commonly as a solitary mass in the mediastinum, but occurrence at extrathoracic locations where lymphoid tissue exists has been well-documented. In a review of 222 cases Iida et al* reported 67% in the mediastinum, 14% in the neck, 4% in the pelvis and retroperitoneum, and 2% in the axilla. Also known as angiofollicular hyperplasia, Castleman disease can be classified into two histopathologic subgroups: the hyaline-vascular and the plasma cell types. The hyaline-vascular type, characterized by hyalinized follicular centers and prominent interfollicular vascular proliferation, accounts for more than 90% of the cases reported, whereas the plasma cell type, in which hyperplastic follicles are separated by sheets of plasma cells, accounts for fewer than 10% of the cases reported.3 No age or sex predilection has been shown.
654 Abramov
et al
Castleman
Disease
in Pregnancy
The hyaline-vascular type, particularly in its localized form, is usually asymptomatic and benign. Because it is a rare disorder, most cases of Castleman disease are diagnosed only postoperatively. A MEDLINE search of the English literature of the years 1966-1997 and a search of the Cumulated Index Medicus (1955-1965) using appropriate key words identified no reports of Castleman disease occuring during pregnancy. Hence, the case presented here is unique in this respect. Although the hyaline-vascular type is generally asymptomatic, this patient presented with second-trimester vaginal bleeding. The patient’s premature delivery may be attributed to the abdominal operation she had undergone 12 weeks earlier, although the relatively long interval between these two events does not support this assumption. The mass was located in the mesocolon, a rare site for Castleman disease. Whether this site is more frequent in pregnancy remains to be answered. Castleman disease should be kept in mind in the differential diagnosis of an abdominal or retroperitoneal mass during pregnancy. Further progress in preoperative diagnosis is required in order to reduce unnecessary surgical interventions in this group of patients.
References 1. Castleman B, Iverson L, Menendez VP. Localized mediastinal lymph node hyperplasia resembling thymoma. Cancer 1956;9:82230. 2. Iida E, Kohno A, Mikami T, Kumekawa H, Akimoto S, Hamano K. Mesenteric Castleman tumour. J Comput Assist Tomogr 1983;7: 338-40. 3. Keller AR, Hochholzer L, Castleman B. Hyaline-vascular and plasma cell types of giant lymph node hyperplasia of the mediastinum and other locations. Cancer 1972;29:670-83.
Reprints
are not
available.
Received Februa y 4, 1997. Received in revised form March Accepted April 7, 1997.
31, 1997.
Copyright 0 1997 by The American College of Obstetricians Gynecologists. Published by Elsevier Science Inc.
Obsfefrics
and
G Gynecology
Address
CASTLEMAN
disease presenting as an abdominal bleeding during the second trimester
DISEASE
reprint
requests
to:
Nicola Carretti, MD Obstetrics and Gynecology Clinica Ostetrica e Ginecologica dell’Universitri Via Paolo Mascagni, 46 I-53100 Siena ltaly
di Siena
Received December 30, 1996. Received in revised form April 14, 1997. Accepted April 21,1997. Copyright 0 1997 by The American College of Obstetricians Gynecologists. Published by Elsevier Science Inc.
and
mass and vaginal of pregnancy.
IN PREGNANCY Yoram Abmmov, MD, Michel Nadjari, MD, Ayula Abrahamov, MD, Constantin Aeinus, MD, and Shaoul 0. Anteby, MD Background: Castleman disease, a rare disorder characterized by benign proliferation of lymphoid tissues, generally presents as a solitary mediastinal mass. We report the first case of this disease during pregnancy. Case: A 32-year-old woman presented with a large abdominal mass and vaginal bleeding during the second trimester of pregnancy. Abdominal ultrasound demonstrated a large, retroperitoneal solid mass of mixed echogenicity and increased vascularity. The patient underwent explorative laparotomy that revealed a mesenteric mass, histologically consistent with Castleman disease of the hyaline-vascular type. The mass was excised completely, and the immediate postoperative course was uneventful, although the patient went into spontaneous preterm labor during the 29th week of pregnancy. Conclusion: Castleman disease should be considered one of the benign etiologies for an abdominal or retroperitoneal mass during pregnancy. (Obstet Gynecol 1997;90:653-4. 0 1997 by The American College of Obstetricians and Gynecologists.) Castleman disease, a rare disorder characterized by benign proliferation of lymphoid tissue,’ usually presents as a localized soft-tissue mass in the neck or mediastinum. We report a case of mesenteric Castleman From fhe Departments of Obstetrics and Gynecology, and Pathology, Hadassah Hebrew University Medical Center, Ein-Kerem, and jrom the Department of Pediatrics, Shaare-Zedek Medical Center, ]erusalem, Israel.
VOL.9O,NO.4,PART
2,OCTOBER
1997
Case A 32-year-old eight, para
married, six, presented
generally healthy woman, gravida with vaginal bleeding during her
15th gestational week. Physical examination revealed a large, semisolid, partially fixed, nontender, irregularly outlined mass above and right of the uterus. There was a small amount of uterine bleeding, but the cervix was closed. Abdominal sonography indicated a single, live fetus consistent with the stage of gestation, with an adequate amount of amniotic fluid and no signs of placental separation. A large solid mass of mixed echogenicity and increased vascularity by color Doppler, 10 X 8 cm in diameter, was demonstrated in the right retroperitoneum, near the lower pole of the right kidney. The patient was hospitalized, and 10 days after vaginal bleeding subsided, she underwent an exploratory laparotomy. There
was
a 12-X-6-cm
solid
mass
near
the
root
of the
transverse mesocolon, adjacent to the superior mesenteric vessels and surrounded by loose connective tissue. No enlarged lymph nodes were palpated, and the genital and abdominal organs were normal. The mass was excised completely. A frozen section suggested a high probability of lymphoma, but the permanent histopathologic sections revealed numerous hyperplastic lymphoid follicles in a structure
of a lymph
node
covered
by a thin
capsule.
Germinal
centers were transfixed by penetrating vessels with foci of hyalinization, whereas mantle zones showed concentric hyperplasia (Figure 1). Interfollicular stroma contained abundant vessels, mature small lymphocytes, and almost no plasma cells. These findings are diagnostic for Castleman disease of the hyaline-vascular type. The patient had an uneventful postoperative course, although
12 weeks
later
she developed
uterine
contractions
and
delivered prematurely during the 29th week of pregnancy. The infant, weighing 1410 g at delivery, was admitted to the neonatal intensive care unit, where he did well and was discharged in good general condition 6 weeks later.
0029-7844/97/$17.00 PII SO'J29-7844(97)00222-6
653
Figure 1. Photomicrograph (X100) showing a lymph follicle composed of concentrically arranged small lymphocytes with a hyalinized germinal center penetrated by a number of small vessels. The interfollicular space is highly vascular and contains almost no plasma cells. These features are characteristic of Castleman disease of the hyalinevascular type.
Cowmen t Castleman disease, first described in 1956 after analysis of a number of cases of enlarged mediastinal lymph nodes that initially had been classified erroneously as thymic tumors,’ is attributed to lymph node hyperplasia of unknown etiology. The disorder is seen most commonly as a solitary mass in the mediastinum, but occurrence at extrathoracic locations where lymphoid tissue exists has been well-documented. In a review of 222 cases Iida et al* reported 67% in the mediastinum, 14% in the neck, 4% in the pelvis and retroperitoneum, and 2% in the axilla. Also known as angiofollicular hyperplasia, Castleman disease can be classified into two histopathologic subgroups: the hyaline-vascular and the plasma cell types. The hyaline-vascular type, characterized by hyalinized follicular centers and prominent interfollicular vascular proliferation, accounts for more than 90% of the cases reported, whereas the plasma cell type, in which hyperplastic follicles are separated by sheets of plasma cells, accounts for fewer than 10% of the cases reported.3 No age or sex predilection has been shown.
654 Abramov
et al
Castleman
Disease
in Pregnancy
The hyaline-vascular type, particularly in its localized form, is usually asymptomatic and benign. Because it is a rare disorder, most cases of Castleman disease are diagnosed only postoperatively. A MEDLINE search of the English literature of the years 1966-1997 and a search of the Cumulated Index Medicus (1955-1965) using appropriate key words identified no reports of Castleman disease occuring during pregnancy. Hence, the case presented here is unique in this respect. Although the hyaline-vascular type is generally asymptomatic, this patient presented with second-trimester vaginal bleeding. The patient’s premature delivery may be attributed to the abdominal operation she had undergone 12 weeks earlier, although the relatively long interval between these two events does not support this assumption. The mass was located in the mesocolon, a rare site for Castleman disease. Whether this site is more frequent in pregnancy remains to be answered. Castleman disease should be kept in mind in the differential diagnosis of an abdominal or retroperitoneal mass during pregnancy. Further progress in preoperative diagnosis is required in order to reduce unnecessary surgical interventions in this group of patients.
References 1. Castleman B, Iverson L, Menendez VP. Localized mediastinal lymph node hyperplasia resembling thymoma. Cancer 1956;9:82230. 2. Iida E, Kohno A, Mikami T, Kumekawa H, Akimoto S, Hamano K. Mesenteric Castleman tumour. J Comput Assist Tomogr 1983;7: 338-40. 3. Keller AR, Hochholzer L, Castleman B. Hyaline-vascular and plasma cell types of giant lymph node hyperplasia of the mediastinum and other locations. Cancer 1972;29:670-83.
Reprints
are not
available.
Received Februa y 4, 1997. Received in revised form March Accepted April 7, 1997.
31, 1997.
Copyright 0 1997 by The American College of Obstetricians Gynecologists. Published by Elsevier Science Inc.
Obsfefrics
and
G Gynecology