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Saudi Journal of Kidney Diseases and Transplantation
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CASE REPORT  
Year : 2012  |  Volume : 23  |  Issue : 4  |  Page : 794-798
Simultaneous hemorrhage in intracranial aneurysms and in renal cyst in a case of polycystic kidney disease


1 Department of Neurosurgery, Narayna Medical College Hospital, Chinthareddypalem, Nellore, Andhra Pradesh, India
2 Department of Surgery, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha, India
3 Department of Radiology, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha, India

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Date of Web Publication9-Jul-2012
 

   Abstract 

We report an unusual case of simultaneous hemorrhage in intracranial aneurysms and in renal cyst in a case of polycystic kidney disease. A 27-year-old gentleman presented with progressive headache and intermittent vomiting of one month duration. Initial computerized tomography (CT) scan and magnetic resonance imaging/angiography revealed a large mass lesion in the right temporal fossa. Over the previous 15 days, he developed progressive weakness in his left upper and lower limbs, and the headache worsened in severity. A repeat of CT scan showed an evidence of aneurysmal bleed and a large temporal lobe hematoma. The patient underwent urgent evacuation of the intracerebral of hematoma and excision of the redundant aneurysmal sac. The patient made excellent recovery in the post-operative period; however, for him, the pain abdomen was persisting. Detailed work-up with contrast-enhanced abdominal CT scan revealed bilateral multiple cysts in the kidneys with evidence of intracystic hemorrhage on the left side. An extensive search of the literature revealed that this kind of presentation has not been reported previously.

How to cite this article:
Agrawal A, Dwivedi S, Singh BR, Banode PJ. Simultaneous hemorrhage in intracranial aneurysms and in renal cyst in a case of polycystic kidney disease. Saudi J Kidney Dis Transpl 2012;23:794-8

How to cite this URL:
Agrawal A, Dwivedi S, Singh BR, Banode PJ. Simultaneous hemorrhage in intracranial aneurysms and in renal cyst in a case of polycystic kidney disease. Saudi J Kidney Dis Transpl [serial online] 2012 [cited 2020 Feb 21];23:794-8. Available from: http://www.sjkdt.org/text.asp?2012/23/4/794/98163

   Introduction Top


Autosomal-dominant polycystic kidney disease (ADPKD) is one of the most common life-threatening, hereditary disorders. [1],[2] It is well known that there is an increase in incidence of intracranial aneurysms in patients with ADPKD, and rupture of aneurysms can cause serious and potentially fatal subarachnoid hemorrhage about a decade earlier in these patients. [4],[6],[7],[8],[9] Spontaneous subarachnoid hemorrhage from a ruptured intracranial aneurysm can be the ini­tial manifestation of the unsuspected ADPKD. [6],[8],[9] We report an unusual case of simultaneous hemorrhage of intracranial aneurysms and re­nal cyst in a case of polycystic kidney disease. An extensive search of the literature revealed that this kind of presentation has not been reported previously.


   Case Report Top


A 27-year-old gentleman presented with progressive headache and intermittent vomi­ting of one month duration. He was investi­gated with computerized tomography (CT) scan and magnetic resonance imaging/angiography that showed a large mass lesion in the right temporal fossa [Figure 1], [Figure 2], [Figure 3], [Figure 4] and [Figure 5]. The imaging features were suggestive of a large thrombosed aneurysm. Over the previous 15 days, he deve­loped progressive weakness on the left upper and lower limbs, and his headache worsened in severity; also, he noticed a mild left flank ab­dominal pain. A repeat CT scan showed an evidence of aneurysmal bleed and a large temporal lobe hematoma with significant peri-lesional edema, mass effect and midline shift [Figure 6].
Figure 1: CT scan brain plain and contrast axial images showing a large hyperdense mass lesion in the right temporal fossa.

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Figure 2: CT scan 3D reconstruction showing the details of the lesion.

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Figure 3: MRI axial images showing an evidence of fresh and old hemorrhages and clot in the sac.

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Figure 4: MR angiogram shows the multiloculated nature of the aneurismal sac.

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Figure 5: Digital subtraction angiography images showing the isolation of the sac from the main circulation; note the pressure effect on the right internal carotid artery.

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Figure 6: Follow-up scan shows the increase in the size of the hematoma.

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On examination the patient was febrile (100°F) and had pallor. There was mild tender­ness on abdominal examination; however, there was no hepatosplenomegaly and bowel sounds were normal in addition to normal rectal examination. Neurologically, he was in altered sensorium (Glasgow Coma Scale - eye opening to pain, localizing to pain and income-prehensible sounds to pain), pupils were bila­terally symmetrical and reacting to light and there was mild left-sided facial weakness and dense hemiplegia of the left upper and lower limbs. Laboratory investigations were within normal limits (except mild leucocytosis).

The patient underwent urgent evacuation of the intracerebral of hematoma and excision of the redundant aneurismal sac. The patient made an excellent recovery during the post-operative period; however, his abdominal pain persisted. An ultrasound of the abdomen showed multi­ple cystic lesions in both kidneys. Detailed work-up with contrast-enhanced abdominal CT scan revealed bilateral multiple cysts in the kidneys with evidence of intracystic hemorrhage on the left side [Figure 7]. The patient did not disclose any history of hematuria or hypertension.
Figure 7: CT scan of the abdomen shows polycystic disease of the kidneys and evidence of intracystic hemorrhage on the left side.

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   Discussion Top


Although the association of intracranial aneurysm and ADPKD has been known for many years in the literature, the natural history of these lesions is not well defined. [8],[9],[13] It has been reported that up to 8% of ADPKD pa­tients have intracranial aneurysms. [3] Rupture of the intracranial aneurysms is an important complication of ADPKD, [1],[11] and it can be one of the most devastating extrarenal manifes­tations of ADPKD that can lead to early death or profound disability. [3] As in our case, many of the ADPKD patients can have a normal renal function and blood pressure. [2] The risk of rupture of the intracranial aneurysm is five-times more frequent in patients with ADPKD than in the general population. [6],[10] In ADPKD, intracystic hemorrhage in the kidney can be suspected clinically and diagnosed by ultra-sonography, CT or magnetic resonance imaging. [5] Our case was unique, where the patient had both rupture of the intracranial aneurysm as well as the hemorrhage in the renal cyst, and, because of the altered sensorium, the diagnosis of the intra-cystic hemorrhage was delayed.

 
   References Top

1.Belz MM, Fick-Brosnahan GM, Hughes RL, et al. Recurrence of intracranial aneurysms in autosomal-dominant polycystic kidney disease. Kidney Int 2003;63:1824-30.  Back to cited text no. 1
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2.Chauveau D, Pirson Y, Le Moine A, Franco D, Belghiti J, Grunfeld JP. Extrarenal manifes­tations in autosomal dominant polycystic kidney disease. Adv Nephrol 1997;26:265-89.  Back to cited text no. 2
    
3.Gibbs GF, Huston J 3rd, Qian Q, et al. Follow-up of intracranial aneurysms in autosomal-dominant polycystic kidney disease. Kidney Int 2004;65:1621-7.  Back to cited text no. 3
[PUBMED]  [FULLTEXT]  
4.Gieteling EW, Rinkel GJ. Characteristics of intracranial aneurysms and subarachnoid haemorrhage in patients with polycystic kidney disease. J Neurol 2003;250:418-23.  Back to cited text no. 4
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5.Ishikawa E, Kudo M, Minami Y, et al. Intra-cystic hemorrhage in a patient of polycystic kidney with renocolic fistula diagnosed by contrast-enhanced ultrasonography. Inter Med 2008;47:1977-9.  Back to cited text no. 5
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6.Kanne JP, Talner LB. Autosomal dominant polycystic kidney disease presenting as sub-arachnoid hemorrhage. Emerg Radiol 2004; 11:110-2.  Back to cited text no. 6
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7.Kubo S, Nakajima M, Fukuda K, et al. A 4-year-old girl with autosomal dominant poly-cystic kidney disease complicated by a rup­tured intracranial aneurysm. Eur J Pediatr 2004;163:675-7.  Back to cited text no. 7
[PUBMED]  [FULLTEXT]  
8.Ono Y, Ohnishi M, Katsumata A, Nishio S, Kawauchi M, Matsumoto Y. Intracranial internal carotid artery aneurysm associated with extra-cranial occlusion of the ipsilateral internal carotid artery in a patient with polycystic kidney disease.[Article in Japanese] No Shinkei Geka 2007;35:1097-102.  Back to cited text no. 8
[PUBMED]  [FULLTEXT]  
9.Ponte KF, Mont'Alverne FJ, Ribeiro EM, et al. Giant aneurysm of the intracavernous internal carotid artery associated with autosomal domi­nant polycystic kidney disease: Case report. [Article in Portuguese] Arq Neuropsiquiatr 2006;64:881-4.  Back to cited text no. 9
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10.Rinkel GJ, Djibuti M, Algra A, van Gijn J. Prevalence and risk of rupture of intracranial aneurysms: A systematic review. Stroke 1998; 29:251-6.  Back to cited text no. 10
[PUBMED]  [FULLTEXT]  
11.Schrier RW, Belz MM, Johnson AM, et al. Repeat imaging for intracranial aneurysms in patients with autosomal dominant polycystic kidney disease with initially negative studies: A prospective ten-year follow-up. J Am Soc Nephrol 2004;15:1023-8.  Back to cited text no. 11
[PUBMED]  [FULLTEXT]  
12.Schrier RW. Optimal care of autosomal dominant polycystic kidney disease patients. Nephrology (Carlton). 2006;11:124-30.  Back to cited text no. 12
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13.Yves P, Dominique C, Vicente T. Management of cerebral aneurysms in autosomal dominant polycystic kidney disease; J Am Soc Nephrol 2002;13:269-76.  Back to cited text no. 13
    

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Correspondence Address:
Amit Agrawal
Department of Neurosurgery, Narayana Medical College and Hospital, Chinthareddypalem, Nellore 524003, Andhra Pradesh
India
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DOI: 10.4103/1319-2442.98163

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]

This article has been cited by
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[Pubmed] | [DOI]



 

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