A case of an infiltrating lymphangiomatous lesion of the mesentery in a 17-year-old girl that involved the head of the pancreas and surrounding retroperitoneal tissue and dissected through the duodenojejunal wall to form multiple polypoid structures in the mucosa is presented. Histologically, the lesion was characterized by cavernous lymphangiomatous spaces with an unusually prominent smooth muscle component in their walls, and it could be designated as lymphangiomyoma. It is believed that the lesion, in spite of its superficial resemblance to lymphangiomyomatosis, is not related to it because the character of the smooth muscle cells, their distribution, their negative reactions to HMB-45, estrogen and progesterone receptors, and a general aspect of the cavernous lymphangioma-like lesion are all evidence against such a possibility. The polypoid presentation of the lesion in the intestinal lumen is unique and adds yet another category of small intestinal polyps.
SunC-CJTangC-KHillJL.Mesenteric lymphangioma: A case report with transmission and scanning electron microscopic studies. Arch Pathol Lab Med104:316–318, 1980.
10.
ViarWNScottWFJrDonaldJM.Mesenteric cavernous lymphangiomata: Brief review and report of two cases. Ann Surg153:157–160, 1961.
11.
GaliferRBPousJGJuskiewenskiSPasquieMGaubertJ.Intra-abdominal cystic lymphangiomas in childhood. Prog Pediatr Surg11:173–238, 1978.
12.
HebraABrownMFMcGeehinKMRossAJ.Mesenteric, omental, and retroperitoneal cysts in children: A clinical study of 22 cases. South Med J86:173–176, 1993.
13.
BurrowsLRudickJ.Lymphangioma of retroperitoneum and mesentery. J Mount Sinai Hosp NY34:45–49, 1967.
14.
CarnogJEnterlineHT.Lymphanglomyoma: A benign lesion of chyliferous lymphatics synonymous with lymphangiopericytoma. Cancer19:1909–1930, 1966.
15.
TaylorJRRyuJColbyTVRaffinTA.Lymphangioleiomyomatosis: Clinical course in 32 patients. New Engl J Med323:1254–1260, 1990.
16.
WolffM.Lymphangiomyoma: Clinicopathologic study and ultrastructural confirmation of its histogenesis. Cancer31:988–1007, 1973.
17.
WayneERBurringtonJDBaileyWCFavaraBECampbellJB.Retroperitoneal lymphangioma: An unusual cause of the acute surgical abdomen. J Pediatr Surg8:831–832, 1973.
18.
KafkaVNovakK.Multicystic retroperitoneal lymphangioma in an infant appearing as an inguinal hernia.J Pediatr Surg5:573, 1970.
19.
EnzingerFMWeissSW.Soft tissue tumors, 2nd ed.Mosby, St. Louis, pp. 489–532, 1988.
20.
RamaniPShahA.Lymphangiomatosis: Histologic and immunohistochemical analysis of four cases. Am J Surg Pathol17:329–335, 1993.
21.
ElliottRLWilliamsRDBaylesDGriffinJ.Lymphangioma of the duodenum: Case report with light and electron microscopic observation. Ann Surg163:86–92, 1966.
22.
BoggonRPPalfreyAJ.The microscopic anatomy of human lymphatic trunks. J Anat114:389–405, 1973.
23.
BassetFSolerPMarsacJCorrinB.Pulmonary lymphangiomyomatosis: Three new cases studied with electron microscopy. Cancer38:2357–2366, 1976.
24.
CorrinBLiebowAAFriedmanPJ.Pulmonary lymphangiomyomatosis: A review. Am J Pathol79:348–367, 1975.
GraySRCarringtonCBCornogJLJr.Lymphangiomyomatosis: Report of a case with ureteral involvement and chyluria. Cancer35:490–498, 1975.
27.
JoliatGStalderHKapanciY.Lymphangiomyomatosis: A clinico-anatomical entity. Cancer31:455–460, 1973.
28.
LackEEDolanMFFinisioJGroverGSinghMTricheTJ.Pulmonary and extrapulmonary lymphangioleiomyomatosis: Report of a case with bilateral renal angiomyolipomas, multifocal lymphangioleiomyomatosis, and a glial polyp of the endocervix. Am J Surg Pathol10:650–657, 1986.
29.
VazquezJJFernandez-CuervoLFidalgoB.Lymphangiomyomatosis. Morphogenetic study and ultrastructural confirmation of the histogenesis of the lung lesion. Cancer37:2321–2328, 1976.
30.
BaldacciMCCattaniMGMagniE.Linfangiomiomatosi ad esordio retroperitoneale. Studio immunoistochimico ed ultrastrutturale di un caso. Pathologica81:453–461, 1989.
31.
BhattacharyyaAKBaloghK.Retroperitoneal lymphangioleiomyomatosis: A 36-year benign course in a postmenopausal woman. Cancer56:1144–1146, 1985.
32.
ChanJKCTsangWYWPauMYTangMCPangSWFletcherCDM.Lymphangiomyomatosis and angiomyolipoma: Closely related entities characterized by hamartomatous proliferation of HMB-45-positive smooth muscle. Histopathology22:445–455, 1993.
33.
Treilleux SallesIVitreyDBergerFBeurletJPatricotLM.Lymphangiomyomatose extra pulmonaire revelee par une enteropathie exsudative d'evolution rapidement fatale. Ann Pathol9:279–283, 1989.
BonettiFChioderaPLPeaMMartignoniGBosiFZamboniGMariuzziGM.Transbronchial biopsy in lymphangiomyomatosis of the lung. HMB-45 for diagnosis. Am J Surg Pathol17:1092–1102, 1993.
36.
WeeksDAChaseDRMalottRLChaseRLZuppanCWBeckwithJBMierauGW.HMB-45 staining in angiomyolipoma, cardiac rhabdomyoma, other mesenchymal processes, and tuberous sclerosis-associated brain lesions. Int J Surg Pathol1:191–197, 1994.
37.
BergerUKhaghaniAPomeranceAYacoubMHCoombesRC.Pulmonary lymphangioleiomyomatosis and steroid receptors: An immunocytochemical study. Am J Clin Pathol93:609–614, 1990.
38.
CagnanoMBenharrochDGeffenDB.Pulmonary lymphangioleiomyomatosis: Report of a case with associated multiple soft-tissue tumors. Arch Pathol Lab Med115:1257–1259, 1991.
39.
HilliardRIMcKendryJBJPhillipsMJ.Congenital abnormalities of the lymphatic system: A new clinical classification. Pediatrics86:988–994, 1990.
