Accepted Manuscript An Unusual Manifestation of Renal Angiomyolipoma: Pulmonary Fat Embolism Nurullah Hamidi, M.D., Evren Suer, M.D., Mehmet Ilker Gokce, M.D., Iskender Alacayir, M.D., Cetin Atasoy, M.D., Duygu Kankaya, M.D., Ayca Kirmizi, M.D., Onur Telli, M.D., Sumer Baltaci, M.D. PII:

S0090-4295(15)00498-7

DOI:

10.1016/j.urology.2015.05.016

Reference:

URL 19242

To appear in:

Urology

Received Date: 5 May 2015 Revised Date:

15 May 2015

Accepted Date: 21 May 2015

Please cite this article as: Hamidi N, Suer E, Gokce MI, Alacayir I, Atasoy C, Kankaya D, Kirmizi A, Telli O, Baltaci S, An Unusual Manifestation of Renal Angiomyolipoma: Pulmonary Fat Embolism, Urology (2015), doi: 10.1016/j.urology.2015.05.016. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

ACCEPTED MANUSCRIPT An Unusual Manifestation of Renal Angiomyolipoma: Pulmonary Fat Embolism

Author Names:

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Nurullah Hamidi M.D.1, Evren Suer M.D.1, Mehmet Ilker Gokce M.D.1, Iskender

Alacayir M.D.2, Cetin Atasoy M.D.3, Duygu Kankaya M.D.4, Ayca Kirmizi M.D.4, Onur

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Telli M.D.1, Sumer Baltaci M.D.1

Ankara University, School of Medicine, Department of Urology, Ankara, TURKEY

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Ankara University, School of Medicine, Department of General Surgery, Ankara,

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Ankara University, School of Medicine, Department of Radiology, Ankara, TURKEY

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Ankara University, School of Medicine, Department of Pathology, Ankara, TURKEY

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Corresponding Author

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Nurullah HAMİDİ M.D.

Ankara University School of Medicine, Department of Urology, Ankara, TURKEY e- mail: [email protected] Tel: +90 553 205 03 07

Fax: +90 (312) 508 21 47

Informed Consent: Written informed consent was obtained from the patient. Conflict of interests: No conflict of interest was declared by the authors.

ACCEPTED MANUSCRIPT Financial Disclosure: The authors declared that this study has received no financial support. Word counts for the abstract: 63

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Word counts for the manuscript text: 200

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Key words: Angiomyolipoma; Kidney; Pulmonary Fat Embolus; Thrombosis.

ACCEPTED MANUSCRIPT Abstract Renal angiomyolipoma(AML) is most common benign renal mesenchymal tumors. AMLs are usually asymptomatic and frequently affects women. Only

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epitheloid variant has malignant potential. Although life-threatening complications related to retroperitoneal bleeding and massive hematuria are possible, it is often detected incidentally. Pulmonary embolism as the first sypmtom is extremely rare. Herein, we present a case of renal AML who admitted with pulmonary embolism

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symptoms.

ACCEPTED MANUSCRIPT A 43-year-old woman admitted to emergency service with respiratory distress, tachycardia and cheast pain.She had no history of trauma and coagulopathy. Arterial blood analysis revealed hypoxia(PaO2=66mmHg),hypocarbia(PaCO2=32mmHg), alkalosis(pH=7,47) and high D-dimer(690mcg/L) level. Doppler ultrasound of lower

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extremity was free of thrombus. However, computed tomography showed embolus in pulmonary arter and right renal pelvic mass(Figure 1). Based on these findings, the patient was diagnosed pulmonary fat embolus due to renal angiomyolipoma(AML).

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Initial management was performed with respiratory support, intravenous

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methylprednisolone 1.5mg/kg/day and hydration. Right nephrectomy and IVC thrombectomy was performed after vena cava filter placement. Macroscopic examination revealed a hilar well-demarcated nodule, 5cm in diameter, with a yellow,hemorrhagic cut surface. Histologically, the diagnosis of AML was confirmed

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and details were mentioned in figure 2. Angiomyolipomas

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complications related to retroperitoneal bleeding and massive hematuria are

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possible.1 Presenting with pulmonary embolism is extremely rare. Previously, cases of pulmonary embolism developing after nephrectomy(due to angimyolipoma) and

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accompanied by deep venous thrombosis, concurrent with huge colon carcinoma were published.2,3,4 In 2009, first case of renal AML presenting with nontraumatic pulmonary fat embolus was published.5 To the best of our knowledge, this is the second case in the english language literature.

REFERENCES

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ACCEPTED MANUSCRIPT 1. Ljungberg B, Hanbury DC, Kuczyk M, et al. European Association of Urology Renal Cell Carcinoma Guidelines. Eur Urol 2007;51:1502-10. 2. Tan YS, Yip KH, Tan PH, et al. A right renal angiomyolipoma with IVC

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thrombus and pulmonary embolism. Int Urol Nephrol. 2010; 42: 305-8. 3. Sandstrom CK, Pugsley J, Mitsumori LM. Renal angiomyolipoma with nontraumatic pulmonary fat embolus.AJR Am J Roentgenol. 2009;192:275-6.

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4. Ban D, Yamamoto S, Kuno H, et al. A case of huge colon carcinoma and right renal angiomyolipoma accompanied by proximal deep venous

Clin Oncol. 2008; 38: 710-4.

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thrombosis, pulmonary embolism and tumor thrombus in the renal vein. Jpn J

5. Sandstrom CK, Pugsley J, Mitsumori LM. Renal angiomyolipoma with nontraumatic pulmonary fat embolus. AJR Am J Roentgenol. 2009; 192:

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W275-6.

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FIGURE LEGENDS

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Fig 1. Computed tomography scan showing embolus, in fat density(-84 HU), in the left lower lobe pulmonary arter, in axial plane(arrow,A) and coronal plane(arrow,B). A 56x54x40mm mass that it is predominantly of fat, was seen to extend into right renal vein and inferior vena cava from renal pelvis, in coronal plane(arrow,C) and axial plane(arrow,D). Fig 2. The tumour was composed of an admixture of mature adipocytes, spindle cells and thick-walled dysplastic blood vessels(A,B). There was no sign of renal

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ACCEPTED MANUSCRIPT parenchyma invasion but renal vein tumour thrombosis was detected in the hilar fat(C). The spindle cells were positive with smooth muscle actin, demsin(D), HMB-45

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and Melan-A(E).

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ACCEPTED MANUSCRIPT

An Unusual Manifestation of Renal Angiomyolipoma: Pulmonary Fat Embolism.

Renal angiomyolipoma (AML) is the most common benign renal mesenchymal tumors. AMLs are usually asymptomatic and frequently affect women. Only epithel...
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