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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 11  |  Issue : 3  |  Page : 124-127

Takayasu's arteritis in a Libyan female


Department of Endocrine, Tripoli Medical Centre (Tripoli University for Medical Sciences, Faculty of Medicine), Tripoli, Libya

Date of Submission20-Jul-2019
Date of Decision17-Aug-2019
Date of Acceptance25-Aug-2019
Date of Web Publication21-Oct-2019

Correspondence Address:
Hawa Juma El-Shareif
The Endocrine Unit, Tripoli Medical Center, Tripoli
Libya
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmbs.ijmbs_40_19

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  Abstract 


Takayasu's arteritis (TA) is a large-vessel vasculitis that involves the aorta and its major branches. Renal arteries are frequently involved, usually with renovascular hypertension. The prevalence of TA in Arabs is low. A study of the epidemiological and clinical features of TA in Arabs included 197 identified patients between 1995 and 2012 and none of them was Libyan. We report a 61-year-old Libyan woman in whom TA manifested with hypokalemia and arterial hypertension. Previous ultrasound showed renal size asymmetry raised the possibility of renal artery stenosis. The diagnosis of TA was confirmed by magnetic resonance angiography, which showed a thickened abdominal aortic wall, occlusions of the left renal artery and left common iliac artery, stenosis of the right common iliac artery, and stenosis of both subclavian arteries. TA is rarely encountered in Arabs. However, the disease must be considered in patients who present with renovascular hypertension, in a context of other autoimmune disorders.

Keywords: Hypokalemia, hypothyroidism, insidious, Libya, renal artery stenosis, Takayasu's arteritis


How to cite this article:
El-Shareif HJ. Takayasu's arteritis in a Libyan female. Ibnosina J Med Biomed Sci 2019;11:124-7

How to cite this URL:
El-Shareif HJ. Takayasu's arteritis in a Libyan female. Ibnosina J Med Biomed Sci [serial online] 2019 [cited 2019 Dec 7];11:124-7. Available from: http://www.ijmbs.org/text.asp?2019/11/3/124/269658




  Introduction Top


Takayasu's arteritis (TA) is a chronic vasculitis, characterized by chronic granulomatous inflammation of the vessel wall of large- and medium-sized arteries with a predilection for the aorta and its major branches as well as the proximal portions of pulmonary, coronary, and renal arteries.[1] It can lead to progressive stenosis, occlusion, or aneurismal transformation, with subsequent significant morbidity and mortality.[1]

The pathogenesis of TA is still unknown; an interplay of genetics and autoimmune factors has been suggested.[2] It has been described in association with autoimmune disorders, such as systemic lupus erythematosus, rheumatoid arthritis, systemic sclerosis, ankylosing spondylitis, granulomatosis with polyangiitis, Crohn's disease, and hyperthyroidism.[2],[3],[4],[5],[6],[7],[8] Although the disease has a worldwide distribution, and it is more prevalent among Eastern and Southeast Asian populations, the prevalence in Japan was 40 cases/million population compared to 4.7–8.0 per million population in the rest of the globe.[2] The clinical manifestation of TA varies depending on the site and severity of vascular involvement.

A study of the epidemiological and clinical features of TA in Arab countries included 197 patients identified between 1995 and 2012. Among Arabs, the renal artery was involved in 20%–50% of the patients and hypertension was present in approximately one-third. None of these patients was from Libya. We report a case of TA diagnosed during medical evaluation of incidentally discovered hypokalemia in a Libyan patient with hypothyroidism.


  Case Report Top


A 61-year-old Libyan female presented with a medical history of hypothyroidism on L-Thyroxine replacement since 2012, vitiligo since 1980, and chronic sinusitis. She has been on regular follow-up at the endocrine clinic. The results of her routine laboratory studies revealed a low serum potassium level. She denied any constitutional symptoms. There was no prior history of arthralgias, myalgias, or any other symptoms, suggestive connective tissue or atherosclerotic peripheral vascular disease. No symptoms of loss of muscle strength, visual impairment, syncopal episodes, vertigo, or abdominal pain after eating were observed. No suggestive symptoms of renal or gastrointestinal Potassium loss were noted. She denied any use of diuretics, laxative, or herbs. Drug history included intake of levothyroxine 75 μg and simvastatin 20 mg once daily. Her average blood pressure (BP) recording at the outpatient clinic was 130/80 mmHg, and she denied taking any antihypertensive medication. She had a history of surgery for carpal tunnel syndrome in 2012. A discrepancy of kidney size was noted on the ultrasound abdomen in 2009. The patient reported that there was difficulty in measuring BP in her right arm. On examination, she looked well, clinically euthyroid, not anemic or jaundiced. Thinning of the scalp hair and vitiligo patches were present [Figure 1]. Her weight was 72.5 kg, height was 151 cm tall, and body mass index was 31.8 kg/m2. Pulses of the left brachial and radial arteries were not palpable, her pulse rate was 82 beats/min, and BP in the right arm was 140/90 mmHg and in the left arm was 110/70 mmHg. The rest of the cardiovascular and respiratory examination did not reveal any abnormalities. The abdomen was soft, lax, with no organomegaly and no renal bruits. Laboratory parameters are presented in [Table 1]. Thyroid and liver function tests were normal. Magnetic resonance angiography revealed stenosis of both subclavian arteries, thickened abdominal aortic wall, occlusions of the left renal artery and left common iliac artery, and stenosis of right common iliac artery [Figure 2]. She was referred to the rheumatology clinic for further evaluation and management.
Figure 1: Clinical features suggestive of associated autoimmune diseases. (a) Thinning of scalp hair, (b) vitiligo patches

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Table 1: Some relevant laboratory parameters of the patient and the laboratory reference ranges

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Figure 2: Magnetic resonance angiography of the aorta, demonstrating stenosis of both subclavian arteries, thickened abdominal aortic wall, occlusions of the left renal artery and left common iliac artery, and stenosis of right common iliac artery

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  Discussion Top


TA was named in honor of Japanese Ophthalmologist Mikito Takayasu (1859–1938), who first reported a case of the disease in 1905.[9] TA is a large-vessel vasculitis that involves the aorta and its major branches. It occurs most commonly in Asia particularly in young females.[2] In 1990, the American College of Rheumatology proposed six criteria for the diagnosis of TA [Table 2]. Three out of six criteria are needed for the diagnosis, with a sensitivity of 90.5% and a specificity of 97.8%.[10] Angiographic classification[11] defined six types of TA based on the anatomic distribution of vascular involvement is summarized in [Table 3]. Angiographic classification correlates with clinical manifestations and prognosis but cannot differentiate active from burned-out lesions. According to the arterial angiography study, our patient belongs to Type V, with the involvement of ascending aorta, aortic arch, and its branches and abdominal aorta and/or renal arteries.
Table 2: The six American College of Rheumatology criteria for the diagnosis of Takayasu's arteritis

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Table 3: The angiographic classification of Takayasu's arteritis defining six types based on the anatomic distribution of vascular involvement

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Although the disease typically begins in the second or third decades of life, it is not uncommon in the extremes of age. Soto et al. from Mexico reported 9% of their patients at diagnosis as aged >40 years; 10.7% of patients studied by Vanoli et al. were >50 years; 4.9% of patients in a French study were aged >60 years.[12],[13],[14] Time lag to diagnosis is approximately 15 months. A delay in diagnosis of 2–11 years is seen in the West.[2] In our patient, the previous ultrasound report of a discrepancy of renal size pointed to a disease onset beyond 10 years, with a chronic, insidious course.

A recent study of the epidemiological and clinical features of TA among Arab populations showed female predominance. The age average at onset in most series was 31.5 years, and the mean delay in diagnosis was 3.5 years (ranged 1.5–4.2 years).[15],[16] A long delay in the diagnosis, was in part due to the low awareness of a relatively rare disease.

Patients can present in an early stage of disease activity with constitutional features, such as fever, nocturnal sweats, weight loss, headache, and malaise; with or without symptoms caused by vascular stenosis, occlusion, or aneurysms; or may diagnose later in the “burnout” or chronic stage.[15]

Clinical manifestations of the disease vary depending on the sites and severity of vascular lesions. In our patient, the incidental finding of hypokalemia triggered re-assessing the patient history, examination, and investigation. Presence of hypokalemia with a discrepancy in renal size, in an older person with a history of hypertension, hypothyroidism, and hyperlipidemia, raises the probability of atherosclerosis as the cause of renal artery stenosis (RAS). The insidious onset of the disease, the absence of the initial acute inflammatory phase of TA, and the rarity of the condition in Libyans may have contributed to the delay in the diagnosis.


  Conclusions Top


TA is a rare condition. Only a few cases were reported in the international literature from Libya.[17] The disease was diagnosed in the burned-out stage during evaluation of an incidental finding of hypokalemia secondary to RAS. Delays in diagnosis can be reduced by carefully searching for unequal or absent upper extremity pulses and by listening for renal bruits in hypertensive patients. The disease must be considered in patients who present with renovascular hypertension, in a context of other autoimmune disorders.

Declaration of patient consent

The author certifies that she obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgments

We would like to thank Dr. Marwa Alezabi for helping in collecting data and both Dr. Nizar Braham and Dr. Alaeddine Sahnoun, for their expert reporting of the MRI angiography.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Compliance with ethical principles

No prior ethical approval is required for single case reports. However, the patient provided consent for publication, as stated above.



 
  References Top

1.
Hotchi M. Pathological studies on Takayasu arteritis. Heart Vessels Suppl 1992;7:11-7.  Back to cited text no. 1
    
2.
Jain S, Pondaiah SK. Takayasu's arteritis: Review of epidemiology and etiopathogenesis. Indian J Rheumatol 2015,10:S22-9.  Back to cited text no. 2
    
3.
Korkmaz C, Zubaroǧlu I, Kaya T, Akay OM. Takayasu's arteritis associated with rheumatoid arthritis: A case report and review of the literature. Rheumatology (Oxford) 2001;40:1420-2.  Back to cited text no. 3
    
4.
Hilàrio MO, Terreri MT, Prismich G, Len C, Kihara EN, Goldenberg J, et al. Association of ankylosing spondylitis, Crohn's disease and Takayasu's arteritis in a child. Clin Exp Rheumatol 1998;16:92-4.  Back to cited text no. 4
    
5.
Ashraf M, Sayyid MK, Arunachalam R, Sequeira A, Veerendra H.K. A rare case of Takayasu's arteritis with hyperthyroidism. J Clin Diagn Res 2011;5:835-7.  Back to cited text no. 5
    
6.
Saxe PA, Altman RD. Takayasu's arteritis syndrome associated with systemic lupus erythematosus. Semin Arthritis Rheumat 1992;21:295-305.  Back to cited text no. 6
    
7.
Kettaneh A, Prevot S, Biaggi A, Stirnemann J, Fain O, Hocqueloux L, et al. Hyperthyroidism in two patients with Crohn disease and Takayasu arteritis. Scand J Gastroenterol 2003;38:901-3.  Back to cited text no. 7
    
8.
Sato Y, Kaji M, Kagiyama S, Nagasawa K. Aortitis syndrome and hyperthyroidism – A case report and review of the Japanese literature. Kurume Med J 1980;27:113-7.  Back to cited text no. 8
    
9.
Numano F. The story of Takayasu arteritis. Rheumatology (Oxford) 2002;41:103-6.  Back to cited text no. 9
    
10.
Arend WP, Michel BA, Bloch DA, Hunder GG, Calabrese LH, Edworthy SM, et al. The American College of Rheumatology 1990 criteria for the classification of Takayasu arteritis. Arthritis Rheum 1990;33:1129-34.  Back to cited text no. 10
    
11.
Moriwaki R, Noda M, Yajima M, Sharma BK, Numano F. Clinical manifestations of Takayasu arteritis in India and Japan – New classification of angiographic findings. Angiology 1997;48:369-79.  Back to cited text no. 11
    
12.
Soto ME, Espinola N, Flores-Suarez LF, Reyes PA. Takayasu arteritis: Clinical features in 110 Mexican mestizo patients and cardiovascular impact on survival and prognosis. Clin Exp Rheumatol 2008;26:S9-15.  Back to cited text no. 12
    
13.
Vanoli M, Daina E, Salvarani C, Sabbadini MG, Rossi C, Bacchiani G, et al. Takayasu's arteritis: A study of 104 Italian patients. Arthritis Rheum 2005;53:100-7.  Back to cited text no. 13
    
14.
Arnaud L, Haroche J, Limal N, Toledano D, Gambotti L, Costedoat Chalumeau N, et al. Takayasu arteritis in France: A single-center retrospective study of 82 cases comparing white, North African, and black patients. Medicine (Baltimore) 2010;89:1-7.  Back to cited text no. 14
    
15.
Kerr GS, Hallahan CW, Giordano J, Leavitt RY, Fauci AS, Rottem M, et al. Takayasu arteritis. Ann Intern Med 1994;120:919-29.  Back to cited text no. 15
    
16.
Mustafa KN. Takayasu's arteritis in Arabs. Clin Rheumatol 2014;33:1777-83.  Back to cited text no. 16
    
17.
Mishra A, Bhaktarahalli JN, Ehtuish EF. Takayasu arteritis diagnosed by 16-row multidetector CT angiography. Neurosciences 2007;12:73-5.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]



 

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