A rare cause of hemorrhagic shock in children: bladder hemangioma

(1)

A rare cause of hemorrhagic shock in children:

bladder hemangioma

Kemal Var

ım Numanoğlu

⁎

, Duygu Tatl

ı

Faculty of Medicine, Department of Pediatric Surgery, Zonguldak Karaelmas University, Kozlu, 67600 Zonguldak, Turkey

Received 14 December 2007; accepted 11 February 2008

Key words: Hemangioma; Hemorragic shock; Child; Gross hematuria; Urinary bladder

Abstract We report the first case of urinary bladder hemangioma that caused hemorrhagic shock consequent to massive hematuria. A 2-year-old girl was presented with hemorrhagic shock and macroscopic hematuria. After rapid resuscitation was performed, radiologic investigations demonstrated a solid enhancing mass originating from the dome of bladder. Partial cystectomy was made. Histopathologic examination of resected specimen revealed cavernous hemangioma. Although bladder hemangiomas are rare benign tumors, it should be kept in mind that they can lead to life-threatening complications when the diagnosis is delayed.

Hemangiomas, the most frequent benign tumors of childhood, are rarely located in the urinary tract. Only 0.6% of bladder tumors are hemangiomas [1]. The typical cause of patient admission is macroscopic hematuria. In the present case report, a urinary bladder hemangioma has been presented that caused hemorrhagic shock consequent to massive hematuria.

1. Case report

A 2-year-old girl was admitted with complaints of malaise and pallor. She had sustained a minor trauma to the abdomen while playing with her sibling, and ever since that incident, her urine color was seen to have darkened. Her parents realized that she was recently pale and weak and had her

admitted to the emergency service when complaints began to appear. There were no significant characteristics in her medical and family histories.

On admission, her general status was poor, and she was lethargic. She had per oral cyanosis, oxygen saturation was 85%, body temperature was 36°C, cardiac pulse rate was 174/min, breathing rate was 40/min, and blood pressure was 60/40 mm Hg. On the general examination, skin and mucosa were cold and pale. Suprapubic tenderness was the only finding. After urinary catheterization, massive hema-turia was observed. On admission, hemoglobin level was 3.6 g/dL; hematocrit was 8%, and platelet count was 462.000/ mm3_{. Complete urinalysis showed abundant erythrocytes.}

She was in hemorrhagic shock with the present findings. Therefore, after central catheterization, transfusion of erythrocyte suspension and appropriate fluid replacement were performed. After an emergent resuscitation was performed, a urinary tract ultrasonography was performed, which revealed findings consistent with hematoma in bladder. On computerized tomography, there was a solid enhancing mass measuring 44 × 37 mm originating from the dome of the urinary bladder (Fig. 1).

⁎ Corresponding author. Tel.: +90 372 2610169x2458; fax: +90 372 2610155.

E-mail addresses:varimk@yahoo.com(K.V. Numanoğlu),

dddtatli@mynet.com(D. Tatlı).

www.elsevier.com/locate/jpedsurg

(2)

After hemodynamic stabilization, she was taken to operation room. On cystotomy, a wide-based, hemorrhagic nodular tumor of 4 × 5 cm size, located on the fundus of the bladder extending through both lateral sides was detected, and partial cystectomy was performed (Fig. 2). Histopatho-logic analysis of the specimen established a diagnosis of cavernous hemangioma.

When no abnormalities were observed in the post-operative period, she was discharged from the hospital. In the interval follow-up, there was no recurrence at the fourth month.

2. Discussion

Hemangiomas, which are tumors of vascular origin growing by cellular proliferation, are the most frequent childhood tumors. Hemangiomas may be located on the skin,

in the viscera, or both. A urinary tract localization is considerably rare, and they are frequently found in kidneys and the urinary bladder, in decreasing order of frequency[2]. Urinary hemangiomas were first defined by Broca in 1869 as an autopsy finding [3]. In the literature, there are about 100 cases that have been reported since 1942 [4]. The most frequent symptom of urinary bladder heman-giomas is painless macroscopic hematuria, which does not cause any other discomfort. Hematuria may cause anemia and suprapubic tenderness in some patients. However, bleeding so marked as to cause a hemorrhagic shock has not been reported in any of the cases studied in the literature. Bladder irritation and urinary retention are findings encountered especially in hemangiomas located at the neck of the bladder.

Bladder hemangioma has been found to occur at any age, with Chen et al [5] reporting 19 patients with this tumor with a mean age of 58 years and a range of 19 to 79 years. However, hemangioma in childhood occurs relatively rarely, with some cases reported as part of a congenital cavernous syndrome such as the Sturge-Weber and the Klippel-Trenaunay-Weber syndromes. Bladder hemangiomas may be concurrently seen with skin hemangiomas [4,5].

Lesions are generally solitary, and they vary 0.5 to 10 cm. In most cases, the tumor is smaller than 3 cm. In 3% of cases, the lesion is superficial and extends to the submucosa only. The muscular layer of the bladder is involved in 64% of cases

[6]. The tumor is most frequently located in the fundus of the bladder, on the posterior wall, or the trigone. They are rarely located on the neck of the vesica. Cavernous hemangioma is the dominant histologic type. However, capillary and arteriovenous hemangiomas are rare[7].

Ultrasonography, computerized tomography, magnetic resonance imaging, and technetium 99-labeled albumin scan are all used to determine the characteristics, location, and extent of the tumor[8,9].

In hemangiomas covering a wide area or those multiple in number, partial cystectomy or bladder augmentation with simple cystectomy, and in small tumors YAG (yttrium aluminium garnet), laser coagulation are the treatment options that may be preferred[10].

Although bladder hemangiomas are rare benign tumors observed in childhood, it should be kept in mind that they can lead to life-threatening complications when the diagnosis is delayed, as in the case presented.

References

[1] Grignon DJ. Neoplasms of the urinary bladder. In: Bostwick DG, Eble JN, editors. Urologic surgical pathology. St. Louis: Mosby-Year Book; 1997. p. 214-315.

[2] Jahn H, Nissen HM. Haemangioma of the urinary tract: review of the literature. Br J Urol 1991;68:113-7.

[3] Susset J, Korzinstone C, Masse S. Cavernous hemangioma of vesical neck. Urology 1981;17:75-6.

Fig. 1 Computerized tomography demonstrated a solid enhan-cing mass (*) measuring 44 × 37 mm originating from the dome of the urinary bladder.

Fig. 2 Intraoperative findings. Active bleeding was seen on the tumor.

(3)

[4] Ikeda T, Shimamoto K, Tanji N, et al. Cavernous hemangioma of the urinary bladder in an 8-year-old child. Int J Urol 2004;11:429-31. [5] Chen M, Lipson SA, Hricak H. MR imaging evaluation of benign

mesenchymal tumors of urinary bladder. AJR Am J Roentgenol 1997;168:399-403.

[6] Hendry WF, Vinnicommbe J. Haemangioma of bladder in children and young adults. Br J Urol 1971;43:309-16.

[7] Cheng L, Nascimento AG, et al. Hemangioma of the urinary bladder. Cancer 1999;86(3):498-504.

[8] Kunimi K, Hisazumi H, Kadaya M, et al. Magnetic resonance imaging of bladder hemangioma. Abdom Imaging 1993;18:97-9.

[9] Bryniak SR, Morales A, Challis T. Assessment of cavernous hemangioma of bladder by Technetium-99-tagged albumin scan. Urology 1979;13:289-90.

[10] Kato M, Chiba Y, Sakai K, et al. Endoscopik neodymium: yttrium aluminium garnet (Nd:YAG) laser irradiation of the bladder heman-gioma associated with Klippel-Weber syndrome. Int J Urol 2000;7:145-8.

E3 Bladder hemangioma in children