Vol. 114, November
THE JOURNAL OF UROLOGY
Printed in U.S.A.
Copyright© 1975 by The Williams & Wilkins Co.
LIPOPLASTIC LYMPHADENOPATHY SIMULATING MALIGNANT LYMPHOMA AND PELVIC LIPOMATOSIS: REPORT OF A CASE AND REVIEW OF THE LITERATURE LARRY G. MANNING, RUSSELL J. PISCHINGER
AND
LEWIS M. BOBROFF
From the Departments of Surgery and Radiology, Wilford Hall United States Air Force Medical Center, Lackland Air Force Base, Texas
ABSTRACT
The second known case of pelvic lipoplastic lymphadenopathy is presented. Roentgenographic studies including lymphangiography were instrumental in the diagnostic evaluation but a histopathologic diagnosis was essential to exclude a neoplastic process. The etiology and natural history of this disease are indeterminant but seemed to pursue a benign 17-year course in our patient. A retroperitoneal lymph node dissection is not indicated unless encroachment on adjacent pelvic viscera causes mechanical obstruction. Lipoplastic lymphadenopathy as originally described by Morehead and McClure is a rare, benign disorder of lymphoid tissue. 1• 2 It represents replacement of lymph follicles with mature fat associated with enlargement of these nodes. Although the enlarged nodes are characteristically found in the axillae, 1 case involving the pelvic and retroperitoneal nodes has been described.• We herein report on a patient with a preoperative diagnosis of lipoplastic lymphadenopathy and discuss the differentiation of this lesion from the more common pelvic lipomatosis and malignant lymphoma. CASE REPORT
A 62-year-old multigravida white woman was hospitalized for evaluation of enlarging pelvic masses known to be present for at least 7 years. She complained only of mild constipation. Significant medical history included: 1) removal of a chocolate cyst of the right ovary 23 years previously, 2) exploratory laparotomy for pelvic masses 17 years ago with operative findings of fatty lymph nodes, 3) right axillary node biopsy 14 years earlier which pathologically showed an encapsulated lipoma with surrounding lymphoid tissue, 4) a total abdominal hysterectomy and bilateral salpingo-oophorectomy 11 years previously for a left adnexal mass that histologically showed only uterine leiomyomas and 5) cholecystectomy and biopsy of an ileocecal lipoma 8 years ago. The presumptive diagnosis was pelvic lipomatosis based on the physical findings and contrast studies of the colon and urinary tract. Because of a questionable increase in the size of the pelvic masses and the rarity of pelvic lipomatosis in women further evaluation was undertaken. The abdominal physical findings were confined to the pelvis. The uterus was absent and there were Accepted for publication May 30, 1975. 788
bilateral, non-tender, firm, fixed adnexal masses approximately 6 by 7 cm. in size. The roentgenologic studies suggested the diagnosis in this patient. The excretory urogram (IVP) showed an abnormal axis of the left kidney with displacement of the lower pole laterally. There was also marked lateral displacement of the proximal third of the left ureter. The right ureter and collecting system appeared normal (fig. 1, A). The bladder was pear-shaped and bilateral extrinsic compression was noted (fig. 1, B). A barium enema showed displacement of the small bowel upward, floor (fig. 2, A). An upper gastrointestinal study showed displacement of the small bowel upward suggesting a low abdominal or pelvic mass (fig. 2, B). Because of the possibility of a retroperitoneal neoplastic process, a lower extremity lymphangiogram was performed. A massively distorted lymphatic system was noted with grossly enlarged and abnormal-appearing pelvic and periaortic lymphatics (fig. 3). Compound B-mode ultrasound scanning of the abdomen demonstrated multiple sonolucent and complex periaortic and pelvic masses. These findings were consistent with those noted in the urogram and barium enema examinations. A total body 67 gallium scintiscan was normal. Despite the patient's good general health and roentgenographic findings suggestive of lipoplastic lymphadenopathy, an exploratory laparotomy was done to exclude a pelvic malignancy or lymphoma. The gross and pathologic findings included massively enlarged lymph nodes along both iliac vessels and extending in the periaortic area to the level of the renal arteries. Frozen section microscopy showed fat with lymph follicles. Permanent histopathology sections showed a large amount of encapsulated fat in the lymph nodes. The fat cells were normal on electron microscopy. No evidence of acute or chronic inflammation was noted.
PELVIC LIPOPLASTIC LYMPHADENOPATHY
789
FIG. 1. A, IVP demonstrates marked lateral deviation of left upper ureter and left lower renal pole by para-aortic mass. B, cystogram demonstrates elongation and bilateral compression of bladder reminiscent of pelvic lipomatosis.
FIG. 2. A, post-evacuation film from barium enema examination reveals elevation of sigmoid colon and elongation of rectosigmoid. B, small bowel examination reveals elevation of small bowel from pelvis by large pelvic mass effect.
F!G. 3. bizarre
film from bilateral lower channels. B, 24-hour of :1on.-Hodgkin's
demonstrates dilated, tortuous o.nd frn?.my pe~v~c 2.nd para-aottic lymph
790
MANNING, PISCHINGER AND BOBROFF
DISCUSSION tion of the left ureter, a finding not described in Lipoplastic lymphadenopathy was first de- classical pelvic lipomatosis. The radiologic appearance in our case was masscribed by Morehead and McClure who noted the sively enlarged retroperitoneal and pelvic lymph excess accumulation of fat within axillary lymph nodes submitted in radical mastectomy speci- nodes confirmed by lymphangiography. Lymphanmens. 1• 2 They considered it a metaplastic rather giographic appearance was not inconsistent with a than an infiltrative process. Our patient had had non-Hodgkin's lymphoma but the negative 67 gala previous axillary lymph node biopsy consistent lium scintiscan and general well being of the pawith this diagnosis. Wolfe! and Smalley reported tient mitigated against it. These findings as well the only previous case involving the pelvis. 3 To our as a history of fatty pelvic masses strongly indiknowledge no cases have been diagnosed preoper- cated lipoplastic lymphadenopathy. Unless mechanical obstruction to adjacent pelatively. The etiology and natural history of lipoplastic vic viscera exists, operative intervention in lipolymphadenopathy are unclear. Whether this is an plastic lymphadenopathy is solely for the purpose abnormal involutional process or a response to of diagnosis. Although the exact natural history of inflammation is unknown. The differential diagno- this disease process is unknown, retrospective sis of this lesion must include pelvic lipomatosis analysis of our patient would seem to indicate that and malignant lymphoma. Metastatic disease the disease pursued a benign course for at least 17 years. Aggressive operative intervention was theremust also be excluded. Pelvic lipomatosis is a rare, benign disease fore not deemed indicated. consisting of an overgrowth of fat surrounding the REFERENCES bladder and rectosigmoid. 4 • 5 Radiologically, it is manifested by radiolucent pelvic densities, eleva- 1. Morehead, R. P. and McClure, S.: Lipoplastic lymphtion and elongation of the bladder, and elevation adenopathy. Amer. J. Path., 29: 615, 195:3. and straightening of the sigmoid colon. In our 2. Morehead, R. P.: Human Pathology. New York: patient the urographic findings in the pelvis simuMcGraw-Hill Book Co., p. 1307, 1965. late a pelvic lipomatosis (although no definite 3. Wolfe!, D. A. and Smalley, R. H.: "Lipoplastic" lymphadenopathy. Amer. J. Roentgen., 112: 610, radiolucency was noted). However, the marked 1971. lateral displacement of the left ureter and kidney 4. Barry, J.M., Bilbao, M. K. and Hodges, C. V.: Pelvic was inconsistent with this diagnosis. In addition, lipomatosis: a rare cause of suprapubic mass. J. although pelvic lipomato~s has been described in Urol., 109: 592, 1973. women, it is found almost exclusively in male 5. Malter, I. J. and Omell, G. H.: Pelvic lipomatosis in a subjects.•· 6 It is noteworthy that the 2 reported woman. A case report. Obst. Gynec., 37: 63, 1971. cases of pelvic lipomatosis in women in which IVP 6. Goldstein, H. M. and Vargas, C. A.: Pelvic lipomatois illustrated demonstrate significant lateral deviasis in females. J. Canad. Ass. Radio!., 25: 65, 1974.
THE JOURNAL OF UROLOGY
Printed in U.S.A.
Copyright© 1975 by The Williams & Wilkins Co.
LIPOPLASTIC LYMPHADENOPATHY SIMULATING MALIGNANT LYMPHOMA AND PELVIC LIPOMATOSIS: REPORT OF A CASE AND REVIEW OF THE LITERATURE LARRY G. MANNING, RUSSELL J. PISCHINGER
AND
LEWIS M. BOBROFF
From the Departments of Surgery and Radiology, Wilford Hall United States Air Force Medical Center, Lackland Air Force Base, Texas
ABSTRACT
The second known case of pelvic lipoplastic lymphadenopathy is presented. Roentgenographic studies including lymphangiography were instrumental in the diagnostic evaluation but a histopathologic diagnosis was essential to exclude a neoplastic process. The etiology and natural history of this disease are indeterminant but seemed to pursue a benign 17-year course in our patient. A retroperitoneal lymph node dissection is not indicated unless encroachment on adjacent pelvic viscera causes mechanical obstruction. Lipoplastic lymphadenopathy as originally described by Morehead and McClure is a rare, benign disorder of lymphoid tissue. 1• 2 It represents replacement of lymph follicles with mature fat associated with enlargement of these nodes. Although the enlarged nodes are characteristically found in the axillae, 1 case involving the pelvic and retroperitoneal nodes has been described.• We herein report on a patient with a preoperative diagnosis of lipoplastic lymphadenopathy and discuss the differentiation of this lesion from the more common pelvic lipomatosis and malignant lymphoma. CASE REPORT
A 62-year-old multigravida white woman was hospitalized for evaluation of enlarging pelvic masses known to be present for at least 7 years. She complained only of mild constipation. Significant medical history included: 1) removal of a chocolate cyst of the right ovary 23 years previously, 2) exploratory laparotomy for pelvic masses 17 years ago with operative findings of fatty lymph nodes, 3) right axillary node biopsy 14 years earlier which pathologically showed an encapsulated lipoma with surrounding lymphoid tissue, 4) a total abdominal hysterectomy and bilateral salpingo-oophorectomy 11 years previously for a left adnexal mass that histologically showed only uterine leiomyomas and 5) cholecystectomy and biopsy of an ileocecal lipoma 8 years ago. The presumptive diagnosis was pelvic lipomatosis based on the physical findings and contrast studies of the colon and urinary tract. Because of a questionable increase in the size of the pelvic masses and the rarity of pelvic lipomatosis in women further evaluation was undertaken. The abdominal physical findings were confined to the pelvis. The uterus was absent and there were Accepted for publication May 30, 1975. 788
bilateral, non-tender, firm, fixed adnexal masses approximately 6 by 7 cm. in size. The roentgenologic studies suggested the diagnosis in this patient. The excretory urogram (IVP) showed an abnormal axis of the left kidney with displacement of the lower pole laterally. There was also marked lateral displacement of the proximal third of the left ureter. The right ureter and collecting system appeared normal (fig. 1, A). The bladder was pear-shaped and bilateral extrinsic compression was noted (fig. 1, B). A barium enema showed displacement of the small bowel upward, floor (fig. 2, A). An upper gastrointestinal study showed displacement of the small bowel upward suggesting a low abdominal or pelvic mass (fig. 2, B). Because of the possibility of a retroperitoneal neoplastic process, a lower extremity lymphangiogram was performed. A massively distorted lymphatic system was noted with grossly enlarged and abnormal-appearing pelvic and periaortic lymphatics (fig. 3). Compound B-mode ultrasound scanning of the abdomen demonstrated multiple sonolucent and complex periaortic and pelvic masses. These findings were consistent with those noted in the urogram and barium enema examinations. A total body 67 gallium scintiscan was normal. Despite the patient's good general health and roentgenographic findings suggestive of lipoplastic lymphadenopathy, an exploratory laparotomy was done to exclude a pelvic malignancy or lymphoma. The gross and pathologic findings included massively enlarged lymph nodes along both iliac vessels and extending in the periaortic area to the level of the renal arteries. Frozen section microscopy showed fat with lymph follicles. Permanent histopathology sections showed a large amount of encapsulated fat in the lymph nodes. The fat cells were normal on electron microscopy. No evidence of acute or chronic inflammation was noted.
PELVIC LIPOPLASTIC LYMPHADENOPATHY
789
FIG. 1. A, IVP demonstrates marked lateral deviation of left upper ureter and left lower renal pole by para-aortic mass. B, cystogram demonstrates elongation and bilateral compression of bladder reminiscent of pelvic lipomatosis.
FIG. 2. A, post-evacuation film from barium enema examination reveals elevation of sigmoid colon and elongation of rectosigmoid. B, small bowel examination reveals elevation of small bowel from pelvis by large pelvic mass effect.
F!G. 3. bizarre
film from bilateral lower channels. B, 24-hour of :1on.-Hodgkin's
demonstrates dilated, tortuous o.nd frn?.my pe~v~c 2.nd para-aottic lymph
790
MANNING, PISCHINGER AND BOBROFF
DISCUSSION tion of the left ureter, a finding not described in Lipoplastic lymphadenopathy was first de- classical pelvic lipomatosis. The radiologic appearance in our case was masscribed by Morehead and McClure who noted the sively enlarged retroperitoneal and pelvic lymph excess accumulation of fat within axillary lymph nodes submitted in radical mastectomy speci- nodes confirmed by lymphangiography. Lymphanmens. 1• 2 They considered it a metaplastic rather giographic appearance was not inconsistent with a than an infiltrative process. Our patient had had non-Hodgkin's lymphoma but the negative 67 gala previous axillary lymph node biopsy consistent lium scintiscan and general well being of the pawith this diagnosis. Wolfe! and Smalley reported tient mitigated against it. These findings as well the only previous case involving the pelvis. 3 To our as a history of fatty pelvic masses strongly indiknowledge no cases have been diagnosed preoper- cated lipoplastic lymphadenopathy. Unless mechanical obstruction to adjacent pelatively. The etiology and natural history of lipoplastic vic viscera exists, operative intervention in lipolymphadenopathy are unclear. Whether this is an plastic lymphadenopathy is solely for the purpose abnormal involutional process or a response to of diagnosis. Although the exact natural history of inflammation is unknown. The differential diagno- this disease process is unknown, retrospective sis of this lesion must include pelvic lipomatosis analysis of our patient would seem to indicate that and malignant lymphoma. Metastatic disease the disease pursued a benign course for at least 17 years. Aggressive operative intervention was theremust also be excluded. Pelvic lipomatosis is a rare, benign disease fore not deemed indicated. consisting of an overgrowth of fat surrounding the REFERENCES bladder and rectosigmoid. 4 • 5 Radiologically, it is manifested by radiolucent pelvic densities, eleva- 1. Morehead, R. P. and McClure, S.: Lipoplastic lymphtion and elongation of the bladder, and elevation adenopathy. Amer. J. Path., 29: 615, 195:3. and straightening of the sigmoid colon. In our 2. Morehead, R. P.: Human Pathology. New York: patient the urographic findings in the pelvis simuMcGraw-Hill Book Co., p. 1307, 1965. late a pelvic lipomatosis (although no definite 3. Wolfe!, D. A. and Smalley, R. H.: "Lipoplastic" lymphadenopathy. Amer. J. Roentgen., 112: 610, radiolucency was noted). However, the marked 1971. lateral displacement of the left ureter and kidney 4. Barry, J.M., Bilbao, M. K. and Hodges, C. V.: Pelvic was inconsistent with this diagnosis. In addition, lipomatosis: a rare cause of suprapubic mass. J. although pelvic lipomato~s has been described in Urol., 109: 592, 1973. women, it is found almost exclusively in male 5. Malter, I. J. and Omell, G. H.: Pelvic lipomatosis in a subjects.•· 6 It is noteworthy that the 2 reported woman. A case report. Obst. Gynec., 37: 63, 1971. cases of pelvic lipomatosis in women in which IVP 6. Goldstein, H. M. and Vargas, C. A.: Pelvic lipomatois illustrated demonstrate significant lateral deviasis in females. J. Canad. Ass. Radio!., 25: 65, 1974.
