47歳男, 左側腹部痛を主訴に入院した.画像診断上, 腎血管筋脂肪腫とその破裂による後腹膜血腫がもっとも疑われ, 迅速診断の上, 血腫除去と摘出術が行われたWe report a case of renal angiomyolipoma with retroperitoneal hemorrhage treated by enucleation in a 47 year-old male. The mass in the anterior side of the left kidney, revealed by sonography and CT, was diagnosed as angiomyolipoma with a retroperitoneal hematoma caused by its spontaneous rupture. Removal of hematoma and enucleation of the tumor were performed after the diagnosis. Diagnosis and treatment of ruptured renal angiomyolipoma are discussed

Ebihara, Kazunori

Kobayashi, Mikio

Kurokawa, Kohei

Kusaba, Teruo

Nakazato, Yohichi

Sugiyama, Sumio

Suzuki, Yoshihiko

Yamanaka, Hidetoshi

Kyoto University Research Information Repository

Title Spontaneous rupture of renal angiomyolipomaAuthor(s)Kurokawa, Kohei; Ebihara, Kazunori; Sugiyama, Sumio;Suzuki, Yoshihiko; Kusaba, Teruo; Nakazato, Yohichi;Kobayashi, Mikio; Yamanaka, HidetoshiCitation泌尿器科紀要 (1991), 37(4): 377-380Issue Date1991-04URL http://hdl.handle.net/2433/117159RightType Departmental Bulletin PaperTextversionpublisherKyoto UniversityActa Urol. Jpn. 37: 377-380, 1991 377 SPONTANEOUS RUPTURE OF RENAL ANGIOMYOLIPOMA Kohei Kurokawa, Kazunori Ebihara, Sumio Sugiyama, Yoshihiko Suzuki and Teruo Kusaba From the Department of Urology, Radiology and Surgery, National Takasaki Hospital Yohichi Nakazato , Mikio Kobayashi and Hidetoshi Yamanaka From the Department of Pathology and Urology, Gunma University School of Medicine We report a case of renal angiomyolipoma with retroperitoneal hemorrhage treated by enuclea-tion in a 47 year-old male. The mass in the anterior side of the left kidney, revealed by sonography and CT, was diagnosed as angiomyolipoma with a retroperitoneal hematoma caused by its sponta-neous rupture. Removal of hematoma and enucleation of the tumor were performed after the diagnosis. Diagnosis and treatment of ruptured renal angiomyolipoma are discussed. Key words: Retroperitoneal hemorrhage, Renal angiomyolipoma, Enucleation INTRODUCTION Renal angiomyolipoma (AML) , which can be easily diagnosed by imaging, is dealt with mainly by conservative treat-ment 1,2) . However, when there is sponta-neous rupture, the kidney is seldom pre-served probably because of certain emergen-cy features in volved3) . We report a case of renal AML accom-panied by spontaneous rupture, a characte-ristic complication , and treated by enu-cleation. We have also reviewed the rele-vant literature. CASE REPORT The patient was a 47-year-old male truck driver. He was admitted to a neighboring hospital for severe left flank pain. He had previously passed a left ureter stone. Sonography indicated a mass on the ante-rior side of the left kidney. The following day he was transferred to our hospital and was suspected of having aneurysmal rup-ture. There was no adenoma sebaceum on the face. The abdomen was swollen, and a mass the size of a child's head was detect-ed along with acute tenderness in the left flank. Decreased intestinal peristalsis was noted . Peripheral blood examination showed leukocytosis (WBC I 5,600/01 m) and anemia (RBC 275 IO/mm, Hb 8.6 g /dl, Ht 26.6%). A biochemical test indicated total protein of 5.4 g/dl, lactic dehydroge-nase, 340IU/I, creatinine, 1.4 mg/dl, blood urea nitrogen , 29.6 mg/dl. Urinalysis showed no abnormalities. Drip infusion pyelography ( DIP) confirmed slight dilata-tion of left renal calices and pelvis and they were slightly compressed from below ( Fig . I, left) . Sonography indicated on the ventral side of the lower pole of the left kidney, an echogenic tumor with a partialy transsonic area at the center as Fig. I. Left: Preoperative DIP shows slight dilatation of left renal calices and pelvis, and their slight compression from below. Right : Postoperative DIP shows no dilatation of rena l calices or pelvis . 378 Acta Urol. Jpn. Vol. 37, No.4, 1991 Fig. 2. Left: Preoperative sonography indicates, on the ventral side of the lower pole of the left kidney, an echogenic mass having a partialy transsonic area at its center as well as a transsonic area extending from the circumference of the tumor to the iliac crest. Right: Selective renal arteriography shows a tortuous coarse, feeding vessels at the area corresponding to the lesion of the lower pole, but there is no indicating bleeding. well as a transsonic area extending from the circumference of the tumor to the iliac crest (Fig. 2, left). Computed tomography (CT) indicated, on the ventral side of the lower pole of the left kidney, a low absorp-tion lesion of about 8 cm in diameter, with a high absorption area extending from the circumference of the lesion to the iliac crest (Fig. 3, A). These findings suggested left renal AML with retroperito-neal hemorrhage due to spontaneous rup-ture. Subsequent selective renal arterio-graphy (A G) showed a tortuous coarse feeding vessel in the area corresponding to the above lesion of the lower pole, but no indication of bleeding. Thus emboliza-tion was not necessary (Fig. 2, right). The anemia was improved by conduct-ing a 5-unit blood transfusion, and the operation was performed ten days after its onset. Laparotomy by left subcostal arched incision indicated a hematoma the size of a child's head along the aorta in the retroperitoneum. After putting a clamp on the renal artery and vein, we exposed the anterior surface of the hema-toma from the outside of the descending colon. It was removed and washed, in two steps. A yellow colored goose-egg size tumor subsequently became evident in the A B Fig. 3. A: Preoperative CT scan indicates, on the ventral side of the lower pole of the left kidney, a low absorption lesion of about 8 em-diameter, having a high absorption area extending from the circumference of the lesion to the iliac crest. B: Postoperative CT scan shows a partial area of insufficient contrast on the lesion, and no residual tumor is evident. lower pole of the left kidney. A portion of the tumor was quickly diagnosed, and AML was confirmed pathologically. The tumor was enucleated. (Ischemic interval, 33 min; blood loss including hematoma, 1,904 ml; operating time, 195 min). Histologically, the tumor tissue consist-ed of adipose tissue, rich in blood vessels, with sporadical smooth muscle cell hyper-plasia perivascularly. Constitutive cells showed neither atypical nor nuclear divi-sional imaging (Fig. 4). Temporarily postoperative ileus occur-red, but was relieved conservatively. Post-operative DIP showed no dilatation and the left renal calices and pelvis were out-lined well (Fig. I, right). CT revealed a area with poor contrast on the ventral side of the lower pole of the left kidney, but no residual tumor (Fig. 3A). Kurokawa, et a\.; Retroperitoneal hemorrhage. Angiomyolipoma 379 Fig. 4. Microscopic appearance of the tumor (left HE, x 44. right HE, x 130). The tumor tissue is composed of adipose tissue, rich in blood vessels, with sporadical smooth muscle cell hyper-plasia perivascularly . Constitutive cells show neither atipical nor nuclear divisional imaging. DISCUSSION An increasing number of cases of renal AML have been reported owing to the improvement in imaging techniques such as CT and sonography. Now we find dif-ficulty in regarding this disease as a very rare one . However, since we occasionally encounter cases which may rupture sponta-neously or can be difficult to be differen-tiated from renal cell carcinoma, this disease still remains to be clinically a very important entity. Spontaneous rupture of renal AML often occurs with flank pain in the affected side , nausea, vomiting, shock and other conditions and is included in the category of the so-called acute abdomen. Therefore, laparotomy is often performed under the preoperative diagnosis of perforated peri-tonitis due to appendicitis4 ) , torsion of ovarian cystS). and others, resulting in nephrectomy. The incidence of spontaneous rupture of renal AML was reported to be 16.5% (32 out of 194 cases ) by Amano et aL3) in Japan and 14.2% ( 15 out of 105 cases ) by Mounded et aL6) in Europe and America. Oestering et aLIl , who classified renal AML according to tumor size, reported an incidence of 90 tumors with hemorrhagic sign out of 178 cases (51 %) and that of 10 out of 75 cases ( 13%) above and below 4 cm . On the other hand, McDougal et aJ.7) reported 78 cases of renal rupture associated with perirenal hematoma. Forty five cases (57.7%) were tumorous, and 26 of them were malignant (including 15 cases of renal cell carcinoma) . They found 9 cases of renal AML among the remaining 19 cases of benign tumor . Therefore, although spontaneous rupture of the tumor is a characteristic finding, renal AML should not be regarded as the main underlying disease , but rather as a malignant tumor that should be given se-rious consideration . In the case of an emergency operation as mentioned here, renal conservation is hardly possible even if renal AML is strongly suspected2,S). This may be mainly because renal AML can not be depicted accurately by the actual ima ge diagnosis, as shown by the existence of those cases which have been difficult to distinguish preoperatively from renal cell carcinoma8 ,g) and because spontaneous rupture itself of a renal tumor is rather among the findings suggestive of the malignancy. We believe that when spontaneous rup-ture of renal AML is strongly suspected by sonography, CT and when the gene-ral condition of the patient is stable , we should try to avoid the emergency operation which is possi ble through such treatments transfusion and others after determining the site of bleeding by arte-riography (embolization lO), if necessary) . After these measures have been taken, we should proceed to the surgery and upon pathological determination of renal AML , should try to conserve the kidney , if pos-sible , by partial nephrectomy4 ) or enuclea-tion lIl , as in the case of renal AML with-out rupture. REFERENCES I ) Oesterling JE , Fishman EK, Goldman SM, et a\.: The management of renal angiomyo-lipoma. J Urol 135: 1121-1124,1986 2 ) Yano S, Morihisa K, Hatanaka Y, et a\. : A case of renal angiomyolipoma treated by enucleation . Rinsho Hinyokika 43: 1079-1078, 1989 3 ) Amano M, Okunobo T, Kawahara H, et a\. : 380 ActaUrol.Jpn.Vol.37,No.4,1991Renalangiomyolipoma:reportoftwocases.Includingspontaneousrupture.ActaUrolJpn30:1813-1825,19844)HoriT,HakariyaHandKidaH:Acaseofangiomyolipomafoundbyretroperitonealbleeding.NishinihonJUrol44:1265-1268,19825)EndoS,KikuchiT,MatsumotoN,etal.:Acaseofspontaneousruptureofrenalan-giomyolipomadetectedbycomputedtomo-graphy.ProgressinAbdominalEmergencyMedicine9:347-350,19896)MoundedIM,ToliaBM,BernieJE,etal.:Symptomaticrenalangiomyolipoma.Reportof8cases,2withspontaneousrupture.JUrol119:684-688,19787)McDougalWS,KurshEDandPerskyL:Spontaneousruptureofthekidneywithperi-renalhematoma.JUrol114:181-184,19758)HiranoS,KobashiK,MikawaI,etal.:Angiomyolipomaofthekidneyindistin-guishablefromrenalcellcarcinomainpre-operativediagnosis:acasereport.Nishi-nihonJUrol48:1885-1888,19869)HartmanDS,GoldmanSM,FriedmanAC,etal.:Angiomyolipoma:ultrasonic-patholo-giccorrelation.Radiology138:451-458,198110)Moorhe dJD,FrikzschePHandHadleyHL:Manage entofhemorrhagesecondarytor nalangiomyolipomawithselectivear-terialembolization.JUrol117:122-123,197711)KawashimaK,KurokawaK,TakahashiH,etal.:Multipleangiomyolipomasofthekid-ney:acasereporttreatedbyenucleation.RinshoHinyokika41:1049-1051,1987(繍 器 鑑 、。,iz27:19901990)和文抄録後腹膜出血にて発症した腎血管筋脂肪種国立高崎病院泌尿器科(医長 ・海老原和典)黒川 公平,海 老原和典国立高崎病院放射線科(医長:鈴木良彦)杉山 純夫,鈴木 良彦国立高崎病院外科(医長:臼井 龍)草 場 輝 夫群馬大学医学部第一病理学教室(主任1石田陽一教授)中 里 洋 一群馬大学医学部泌尿器科学教室(主任:山中英寿教授)小林 幹男,山 中 英寿後腹膜出血を伴 う腎血管筋脂肪腫にたい して,核 出術を行いえた1例を報告した。症例は47歳男性で,左側腹部痛を主訴に入院した.画像診断上,腎 血管筋脂肪腫とその破裂による後腹膜血腫 が もっとも疑われた.迅速診断の上,血 腫除去と核出術が行われた.破裂を伴 う腎血管筋脂肪腫の診断 ・治療等につき考察 した.(泌尿系己要37:377-380,1991)

後腹膜出血にて発症した腎血管筋脂肪種

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後腹膜出血にて発症した腎血管筋脂肪種

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