ࡱ> )+$%&'(c bjbj .x[\x[\>N N $<c":PPPP+++4c6c6c6c6c6c6c$fhFZc]+++++ZcPPc%`%`%`+@PP4c%`+4c%`%`:b,bP.4*\Xb cc0cb i%`ibib4%`+++ZcZc%`+++c++++i+++++++++N > : Ruptura angiomiolipoma bubrega / Renal angiomyolipoma rupture Autori: Ivana Rakin dr.med.1, dr.sc. Dalibor `imunovi dr.med.1,2 Ustanove: Klini ki bolni ki centar Osijek, Zavod za urologiju, Osijek, Hrvatska Medicinski fakultet Osijek, Sveu iliate Josipa Jurja Strossmayera Osijek, Osijek, Hrvatska Autor za dopisivanje: Ivana Rakin, Brae Radia 52, 31550 Ladimirevci-Valpovo,  HYPERLINK "mailto:ivana.rakin@gmail.com"ivana.rakin@gmail.com, 00 385 98 9375199 Skraeni naziv lanka: Angiomiolipom Izjava Izjavljujemo da ne postoji sukob interesa autora prema injenicama prezentiranim u lanku niti osobama. Ivana Rakin Dalibor `imunovi Sa~etak Angiomiolipom je dobroudni tumor bubrega sa dvije podvarijante. U bolesnika sa tuberoznom sklerozom je uglavnom manji i bilateralan, a u drugom obliku dolazi u bolesnika bez tuberozne skleroze kada je uglavnom vei i unilateralan. Lije enje angiomiolipoma ovisi o veli ini tumora i prisustvu simptoma (bolovi, krvarenje). Na elno u angiomiolipoma kod kojih su prisutni umjereni ili izra~eni simptomi i/ili u angiomiolipoma veih od 4 cm mo~e se razmatrati kirurako lije enje. Ipak, u veini slu ajeva se provodi konzervativni tretman praenjem sa redovitom procjenom veli ine (UZV, CT). Zabilje~ene su i iznimno rijetke maligne podvarijante angiomiolipoma, uglavnom u veih tumora. Svaki angiomiolipom mo~e potencijalno rupturirati i biti uzrokom krvarenja koje mo~e biti i ~ivotno ugro~avajue. Rupture su eae u veih angiomiolipoma (>10 cm), a zabilje~ene su i rupture angiomiolipoma povezane sa lokalnom traumom lumbalne regije. Prikazat emo slu aj dvije bolesnice od 37 i 45 godina sa rupturom angiomiolipoma bubrega veli ine 10 cm i posljedi nim retroperitonealnim hematomima veli ine 12 x 9 i 15 x 10 cm. Iako anamnesti ki i klini ki nije zabilje~ena trauma koja je prethodila rupturi, jedna bolesnica navodi dizanje te~eg tereta i klini ki ima kontuzijski otisak u predjelu lijevog kuka, a u druge bolesnice postoji anamnesti ki podatak samo o dizanju teakog tereta bez znakova traume. U injena je hitna eksploracija sa posljedi nom evakuacijom hematoma i nefrektomijom odgovarajueg bubrega. Klju ne rije i: bubreg, angiomiolipom, krvarenje Abstract Angiomyolipoma of the kidney is benign tumor that can be found in two variants. In cases related to tuberous sclerosis is usually smaller and bilateral, while in common variant it comes in patients without genetic predisposition, thus being larger and unilateral. Treatment of angiomyolipoma depends on tumor size and symptoms (bleeding, pain). In angiomyolipoma that are larger then 4 cm and/or have mild to sever symptoms surgical treatment can be considered (embolization, partial nephrectomy or nephrectomy). In majority of cases conservative treatment is advised with regular follow up (ultrasound or CT). In larger angiomyolipoma even extremely rare variants with malignant alteration have been described. Nevertheless, every angiomyolipoma, no matter the size can be place of potential life threatening complication rupture, that comes with extensive bleeding. Ruptures are more common in larger tumors (> 10 cm), but ruptures with correlation to even minor trauma to lumbar region have been described. In this case report we will present two patients (females, 37 and 45 years old), with ruptures of angiomyolipomas larger then 10 cm and consecutive haematomas measuring 12 x 9 cm and 15 x 10 cm, retrospectively. Although, no history of trauma was noted in both cases, both patients claim lifting heavy objects and in one patient contusion print was noted on left hip. In both cases, due to clinical presentation, emergency exploration with evacuation of haematoma and nephrectomy was performed. Keywords: kidney, angiomyolipoma, bleeding Uvod Angiomiolipom bubrega je benigni tumor koji je sastavljen od masnog tkiva, glatko miainih stanica i krvnih ~ila (1). Angiomiolipomi bubrega se pojavljuju u dva razli ita klini ka oblika: kao sporadi ni oblik i u sklopu nasljednih poremeaja. Sporadi ni oblik angiomiolipoma je uglavnom solitaran i unilateralan tumor. U dva nasljedna poremeaja: tuberoznoj sklerozi i limfangioleiomiomatozi angiomiolipom bubrega se pojavljuje u multiplom i esto bilateralnom obliku (2). Patohistoloaki su opisana dva oblika angiomiolipoma: standardni oblik (masne stanice, glatko miaine stanice i krvne ~ile) i epiteloidna varijanta koja osim standardnih stanica sadr~i i velike epiteloidne perivaskularne stanice (3,4). Potonja varijanta esto radioloaki, pa i histoloaki pokazuje osobine karcinoma svijetlih stanica bubrega i predstavlja posebno te~ak klini ki problem (5). Incidencija angiomiolipoma u opoj populacije je oko 0.1% u muakaraca i oko 0.3% u ~ena. Angiomiolipom bubrega se uglavnom utvrdi UZV- nim pregledom u sklopu obrade bolesnika zbog drugih bolesti ili simptoma, tijekom sistematskih pregleda. Vrlo esto su asimptomatski, a isto je povezano sa veli inom angiomiolipoma. Angiomiolipomi manji od 4 cm uglavnom su asimptomatski, dok se kod angiomiolipoma veih od 4 cm pojavljuje rizik od krvarenja, a povezan je sa formiranjem arteriovenskih fistula u parenhimu tumora koje predstavljaju mjesta potencijalne rupture (6). Rizik je najvei u tumora promjera veeg od 10 cm, isti se nazivaju gigantskim angiomiolipomima i potrebna je intervencija bez obzira na odsustvo simptoma. Simptomi angiomiolipoma se dijele u dvije skupine: krvarenje (spontano krvarenje  Wunderlich sindrom, traumatsko krvarenje i krvarenje u trudnoi) i na simptome kroni nog zatajenja bubrega uslijed rasta tumora koji uniatava normalan parenhim bubrega (7,8). Lije enje angiomiolipoma naravno ovisi o veli ini tumora i simptomima. U lije enju je po~eljno dr~ati se na ela  nefron poatedne kirurgije . Selektivna arterijska embolizacija je najbolja metoda u lije enju (9), po mogunosti prije nego ato se dogodi krvarenje, a osim embolizacije primjenjuje se parcijalna nefrektomija. Ponekad, naj eae u slu aju opse~nog krvarenja praenog stalnim padom krvne slike i hemodinamski nestabilnim bolesnikom, mora se u initi i hitna nefrektomija. Prikazom ova dva slu aja ~elimo ukazati na potencijalno ~ivotno ugro~avajuu komplikaciju angiomiolipoma uz osvrt na povezanost traume sa rupturom angiomiolipoma. Prikaz slu aja Bolesnica J.N., 37 godina, do sada zdrava, nije lije ena bolni ki, bez dosadaanje uroloake povijesti i bez stalne terapije zaprimljena u hitnu slu~bu podru ne bolnice. Simptomi pri prijemu su bolovi lijevo lumbalno koji su po eli 12 sati prije prijema, bez povraanja i mu nine, urednog mokrenja, bistrog urina i uredne svijesti. Bolovi su po eli nakon ato je tijekom obavljanja kuanskih poslova podigla te~ak teret, a iako negira traumu po prijemu i tijekom cijelog lije enja na lijevom kuku je vidljiv kontuzijski otisak. Pri dolasku hemoglobin je 117 mg/dl, tlak 130/80 mmHg, koagulogram uredan, urin na postavljeni kateter je bistar i bolesnica je hemodinamski stablina. U injen je hitni UZV abdomena i retroperitoneuma koji je ukazao na veliki hematom u lijevom retroperitoneumu. Na hitnom CT se prikazuje angiomiolipom lijevog bubrega sa opse~nim retroperitonealnim hematomom veli ine 15 x 10 cm (Slika 1). Bolesnica je premjeatena u naau ustanovu i po dolasku je hemodinamski stabilna. Radiolog KBC Osijek telemedicinom potvruje nalaz radiologa iz podru ne ustanove (Slika 2.). Zapo eta je konzervativna terapija, ali bolesnica postaje hemodinamski nestabilna uz pad hemoglobina (94 mg/dl). Odlu ujemo se za hitnu eksploraciju. Tijekom zahvata se ustanovi veliki hematom lijevog retroperitoneuma koji potiskuje crijeva sa bubregom kojem nedostaje gornji pol uz prisutno jako krvarenje. U injena je nefrektomija i daljnji tijek lije enja bolesnice je bio rutinski. Patohistoloaka analiza je potvrdila angiomiolipom gornjeg pola lijevog bubrega sa rupturom. Bolesnica N.N., 45 godina, zaprimljena u hitnoj slu~bi u nonim satima uz kratku anamnezu bolova u desnoj lumbalnoj lo~i koji su po eli nakon ato je naglo podigla dijete. Mokri uredno, pri svijesti je, ali ubrzanog pulsa, oznojena, blijeda, sa tlakom 90/60 mmHg, klini kim pregledom se utvruje palpabilna masa u desnom hemiabdomenu koja je bolna, bez drugih znakova traume. Laboratorijska analiza pokazuje hemoglobin ni~i od 80 mg/dl. Hitni CT pokazuje opse~an hematom desno retroperitoneuma veli ine 12 x 9 cm. Uz suportivnu terapiju tijekom dijagnosti ke obrade bolesnica je i dalje hemodinamski nestabilna. U ini se hitna eksploracija desnog retroperitoneuma tijekom koje se odstrani veliki hematom. Bubreg pokazuje znakove rupture u donjem dijelu sa pukotinom koja se airi prema hilusu bubrega i krvnim ~ilama bubrega. U ini se nefrektomija. Daljni tijek lije enja bolesnice je bez osobitosti i otpuatena je kui dobrog opeg stanja. Patohistoloaka analiza je uz imunohistokemijsko bojanje potvrdila angiomiolipom donjeg pola desnog bubrega veli ine oko 6 cm sa znakovima rupture. Rasprava Angiomiolipom z|    X Z \ P R T ~     $ & rtHJZֻ谻hj('5CJOJQJ^JaJhj('CJOJQJ^JaJ&hj('0J#CJOJQJ^JaJmHsHjhj('Ujhj('U%hj('CJH*OJQJ^JaJmHsH"hj('CJOJQJ^JaJmHsHhj('%hj('5CJOJQJ^JaJmHsH2|~  Z \    & t$dd` $$da$d^)d) & Fd)d^dtJ\23R?GGW`cfshs|sjvlvnv~v d`gd/M$dd d`gd8Z\^ @Ghk**+..//22333P?R?GGGGHPVVWWXXTZVZ]]``cߤߤߤh:TVCJOJQJaJUhj('CJOJQJaJh:TVCJOJQJ^JaJ hj('5CJOJQJ\^JaJh/MCJOJQJ^JaJh8CJOJQJ^JaJhj('CJOJQJ^JaJhj('5CJOJQJ^JaJhj('6bubrega je rijedak benigni tumor, neato eai u ~ena (0,3% poulacije). Angiomiolipom bubrega mo~e biti sporadi ni, kad je uglavnom vei i unilateralan ili u sklopu nekih genetskih poremeaja kad je manji, viaestruk i bilateralan (1). Angiomiolipom je graen od masnog tkiva, krvnih ~ila i glatkog miaia, ovisno o zastupljenosti pojedinih elemenata opisane su i razli ite varijante angiomiolipoma (1,3). Zabilje~ene su i maligne varijante angiomiolipoma koje su eae u veih angiomiolipoma, naj eae oponaaajui cisti ni karcinom svijetlih stanica bubrega (5). Takoer va~no je za napomenuti da je angiomiolipom hormonski ovisan tumor, jer su pojedina istra~ivanja dokazala prisustvo receptora za progesteron i estrogen na epitelnim stanicama, koje mogu biti viae ili manje izra~ene (1). Zbog toga je iznimno va~no pratiti trudnice sa prije dokazanim angiomiolipomom za vrijeme trudnoe, jer su zabilje~ene rupture i jako malih angiomilipoma u trudnoi, vjerovatno uzrokovane visokim razinama hormona (1). Veina angiomiolipoma je asimptomatska, dijagnoza se postavlja naj eae slu ajnim pregledom ultrazvukom zbog nekog drugog razloga. Angiomiolipomi uzrokuju bol i ako se dogodi ruptura, mogu biti razlogom krvarenja u retroperitoneum ili trbuanu aupljinu koje mo~e biti po ~ivot ugro~avajue (4,7,8). Rupture angiomiolipoma mogu biti spontane ili traumatske (10,11). Ruptura angiomiolipoma spontana ili traumatska je eaa u velikih tumora, tako da angiomiolipomi manji od 4 cm uglavnom ne rupturiraju spontano, uz izuzetak trudnica. Pojedina istra~ivanja su uzro no-posljedi no povezala traumu lumbalne regije sa rupturom angiomiolipoma, ak i kad te traume nisu bile zna ajne, ponekad uz odmak od nekoliko dana do rupture angiomiolipoma. Zabilje~ena je i trauma angiomiolipoma nakon fizikalnog pregleda lije nika u bolnici (11). Spontane rupture angiomiolipoma su eae u veih tumora, a dokazano je povezano sa razvojem arteriovenskih fistula u tkivu bubrega koje se formiraju tijekom rasta tumora i predstavljaju mjesto rupture angiomiolipoma (6). Lije enje angiomiolipoma ovisi o veli ini tumora, broju tumora, lokalizaciji tumora, simptomima i bolesniku. U proalosti se veina angiomiolipoma, ako su bili simptomatski ili sa krvarenjem, lije ila nefrektomijom odgovarajueg bubrega. Kasniji razvoj saznanja i tehni kih mogunosti je doveo do primjene parcijalne nefrektomije, ato se odr~alo i do danas. Ipak selektivna embolizacija angiomiolipoma, asimptomatskog ili simptomatskog, je danaanja metoda izbora u lije enju (9). Bolesnice koje su lije ene u naaoj ustanovi uglavnom odgovaraju tipi nom prikazu simptomatskog angiomiolipoma. Kod obje bolesnice anamnesti ki postoji podatak o podizanju te~eg tereta 12 ili viae sati prije prijema, ato je opisano kao mogui razlog rupture angiomilipoma zbog naglog rasta intrabdominalnog tlaka. Klini ki kod bolesnica nije utvreno znakova traume, niti su je navodile, ali kod jedne bolesnice je bio vidljiv kontuzijski otisak na lijevom kuku za koji nije imala objaanjenje. Na~alost, kako selektivna embolizacija nije u tom trenutku bila dostupna kao metoda lije enja u naaoj ustanovi i uzevai u obzir klini ko stanje obje bolesnice, u injena je hitna eksploracija i nefrektomija. Patohistoloaka analiza i imunohistokemijska bojanja su dokazala angiomiolipome u odstranjenih bubrega. Ipak, postavljaju se dva pitanja vezana uz naae bolesnice. Prvo, kako su angiomiolipomi veli ine oko 10 i 6 cm ostali nezabilje~eni do sada, ako uzmemo u obzir dostupnost ultrazvuka u danaanje vrijeme i sveprisutne sistematske preglede, naro ito kod ~ena koje su rodile i vjerovatno imale angimiolipome du~i niz godina? Najjednostavnije je objaanjenje ono koje su ponudile obje bolesnice: do tog trenutka nisu bile bolesne i osim ginekoloakog ultrazvuka nisu bile lije ene niti obraivane. Drugo je pitanje: u ova dva slu aja jesu li ovo bile spontane ili traumatske rupture angiomiolipoma? Odgovor na ovo je te~i. Iako su kod obje bolesnice angiomiolipomi bili vei od 4 cm, ipak postoji podatak o "traumi" ili barem ja em fizi kom naprezanju prije rupture, a u jedne bolesnice i kontuzijski otisak. Uzevai u obzir da je dokazano da i najmanja trauma, ak i pregled abdomena od strane lije nika, mo~e biti uzrokom rupture angiomiolipoma, najlogi nije je objaanjenje da ipak u ovim slu ajevima mo~emo govoriti o "traumatskim" rupturama angiomiolipoma buberga. S obzirom na veli inu angiomiolipoma u obje bolesnice, s vremenom, spontane rupture opisanih angiomiolipoma bi bile o ekivane. Zaklju ak Angiomiolipom bubrega je rijedak benigni tumor bubrega koji u veini slu ajeva ne uzrokuje probleme i mo~e se konzervativno pratiti. U bolesnika u kojih se dogodi ruptura angiomiolipoma, a na istu treba posumnjati u svakog bolesnika sa prije utvrenim angiomiolipomom, traumom u anamnezi i specifi nim simptomima, potrebno je u initi selektivnu embolizaciju angiomiolipoma. Zahvala Dr. I. Brigi (Odjel za urologiju) i dr. A. Borov ak (Odjel za radiologiju) iz OB Virovitica na podacima potrebnim za izradu ovog lanka. Literatura 1. Bissler JJ, Kingswood JC. Renal angiomyolipomata. Kidney International. 2004;66:924-34. 2. Chen P, Jin L, Yang Y, Chen Z, Ni L, Yang S, Lai Y. Giant renal angiomyolipoma: A case report. Molecular and clinical oncology. 2017;7:298-00. 3. Koo KC, Kim WT, Ham WS, Lee JS, Ju HJ, Choi YD. Trends of presentation and clinical outcome of treated renal angiomyolipoma. Yonsei Med J. 2010;51(5):728-34. 4. Unlu C, Lamme B, Nass P, Bolhuis HW. Retroperitoneal haemorrhage caused by a renal angiomyolipoma. Emerg Med J. 2006;23:464-65. 5. Wood A, Young F, O Donnell M. Angiomyolipoma with epithelial cyst masquerading as a cystic renal cell carcinoma. Curr Urol. 2015;9:209-11. 6. Yamakado K, Tanaka N, Nakagawa T, Kobayashi S, Yanagawa M, Takeda K. Renal angiomyolipoma: Relationship between tumor size, aneurysm formation and rupture. Radiology. 2002;225:75-82. 7. Wang HB, Yeh CL, Hsu KF. Spontaneus rupture renal angiomyolipoma with hemorrhagic shock. Inter Med. 2009;48:1111-12. 8. Abdellaoui A, Al-Daraji W, Natarajan V, Sandilands D. Spontaneous renal haemorrhage in the peritoneal cavity. Postgrad Med J. 2006;82:e14. 9. Li D, Pua BB, Madoff DC. Role of embolization in the treatment of renal masses. Semin Intervent Radiol. 2014;31:70-81. 10. Sotoaek S, Marki D, `panjol J, Krpina K, Kne~evi S, Mari i A. Bilateral Wunderlich syndrome caused by spontaneous rupture of renal angiomyolipomas. Case Reports in Urology. 2015;doi:10.1155/2015/316956. 11. Zengin S, Al Behcet, Yildrim C, Oktay MM; Yilmaz DA. Rupturing of renal angiomyolipoma due to physical examination. BMJ Case Reports. 2012;doi:10.1136/bcr.12.2011.5404. Legenda Slika 1. CT sa kontrastom pokazuje defekt gornjeg pola lijevog bubrega Picture 1. Contrast CT showing absence of upper pole of left kidney Slika 2. Hematom retroperitoneuma veli ine 15 x 10 cm Picture 2. Large retroperitoneal haematoma measuring 15 x 10 cm Slika 1. / Picture 1. 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