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Table 1 Review of cases of Takayasu arteritis with pyoderma gangrenosum in the literature

From: Takayasu arteritis with pyoderma gangrenosum: case reports and literature review

Authors, country, year

Sex

Racial or ethnic group

Location of PG

Age at onset of the first manifestations, years

Therapy

Systemic symptoms

First vascular manifestation

Vascular involvement

Other complications

Systemic

Cutaneous

Vascular

Okamura et al. [6] Japan, 2017

M

Japanese

First in left buccal region and then spread to cheek, trunk and lower legs

33

37

33

Betamethasone 1 g/d and tacrolimus 3 mg/d for TA, prednisolone 50 mg/d for PG

Fever

Left cervical pain followed by dizziness

  

Vettiyil et al. [7], India, 2017

F

Indian

Dorsum of left foot

8

10

8

Prednisolone (1 mg/kg/d) and mycophenolate mofetil for TA and PG

 

Pain andclaudication pain over legs and back with breathlessness on exertion

Dilation of the ascending aorta, the left common carotid artery, left subclavian, right subclavian and axillary were narrowed at the origin and proximal portion

Chronic recurrent multifocal osteomyelitis

Loetscher et al. [3], Switzerland, 2016

F

Caucasian

Lower extremities

No record

42

43

Prednisone 50 mg/d, cyclosporine, methotrexate

  

Proximal occlusion of the left subclavian artery, wall thickening of the aortic arch and the subclavian artery

Erythema nodosum

Barrera-Vargas et al. [2], Mexico, 2015

F

Mexican

First in lower limbs and then to head, neck, upper and lower extremities

20

20

26

Prednisone 1.5 mg/kg/d and thalidomide 200 mg/d for PG, three methylprednisolone pulses (1 g/d) and prednisone 1 mg/kg/d and methotrexate 15 mg/week for TA

Fever

Sudden amaurosis in left eye

Both the carotid and vertebral system, all the supra-aortic branches, right pulmonary artery

CT revealed a cystic lesion with an air-fluid level in the lower right pulmonary lobe

Barrera-Vargas et al. [2], Mexican, 2015

M

Mexican

Neck, trunk, upper and lower extremities

25

17

26

Prednisone and cyclosporine, and later thalidomide 200 mg/d, dapsone 100 mg/d, azathioprine 100 mg/d for PG. Prednisone 1 mg/kg/d, thalidomide 200 mg/d, dapsone 100 mg/d, azathioprine 100 mg/d for TA

No

Light-headedness and syncope

Aortic dilatation, narrowing thickening of the wall of the left carotid artery, and celiac trunk stenosis, as well as thickening at the origin of the superior mesenteric artery and left renal artery

Pulmonary nodules,histopathologic analysis showing granulomatous chronic inflammation with giant cells

Futaki et al. [8], Japan, 2009

F

Japanese

Neck, face and forearm

28

28

29

Prednisolone 30 mg/d

Intermittent fever, sore throat and cough

 

Pulmonary artery

TA without typical symptoms

Minagawa et al. [9], Japan, 2009

F

Japanese

Left lower leg

21

16

21

Tacrolimus for PG, prednisolone 25 mg/d for TA and necrotizing vasculitis

Episodic general fatigue and low-grade fever

Dysaesthesia in arms

Stenosis and thickened walls in the subclavian, left carotid and right renal arteries

Necrotizing vasculitis with lobular panniculitis in the deep dermis

Ghosn et al. [10], Lebanon, 2008

F

Lebanese

Face, trunk and extremities

7

7

7

Solumedrol 0.5 mg/kg/d, followed by infliximab 100 mg at weeks 0, 2, and 6

 

Acute-onset right wrist drop

Marked aneurysmal dilatation of the ascending aorta with occlusion of the left and right subclavian arteries, in addition to a large aneurysmal pouch in the right axillary area compressing the nerve plexus

Diagnosed as RP for 5 years

Aoussar et al. [11], France, 2007

F

Caucasian

Forearm

No record

21

23

Prednisone

    

Kanaemistu et al. [12], Japan, 2005

F

Japanese

Neck, chest and arm

No record

34

34

Prednisolone and cyclosporine A, surgery

  

Aneurysm from the ascending aorta to the aortic arch

Thoracic aortic aneurysm

Ujiie et al. [13], Japan, 2004

F

Japanese

First limited to lower legs and then extended to buttocks, lower trunk, upper limbs and scalp

21

29

21

Prednisolone 20 mg/d for TA, prednisone 50 mg/d, cyclosporine 6 mg/kg/d

Hypertension and dizziness

Diminished arterial pulse intensity and bruits

Stenosis in the right subclavian, right vertebral and right carotid arteries and occlusions in the left subclavian and left carotid arteries

 

Fearfield et al. [14], U.K., 1999

F

Caucasian

Perineum, forearms and upper arms

27

27

33

Prednisone 150 mg/d, intravenous immunoglobulin, minocycline 200 mg/d, later methotrexate 7.5 mg/week and cyclosporine 5 mg/kg/d

Mild influenza-

like symptoms, including general muscle aches

Heaviness in arms

Bilateral proximal subclavian artery occlusions with slight narrowing of the right vertebral artery origin

 

Dagan et al. [15], Israel, 1995

M

Israeli

Face and extremities

4

9 months

4

Prednisone 2 mg/kg/d and dapsone 5 mg/kg for PG, high-dose prednisone and surgery for TA

Restless, tired and febrile

Absence of both radial pulses

Severe aneurysmatic dilation of the ascending aorta, severe stenosis of the left subclavian artery

Sterile osteomyelitis

Fullerton et al. [16], 1991

F

Saudi Arabian

Abdomen, followed by rapid spread to legs

No record

28

No record

Prednisone, cyclosporine

    

Frances et al. [4], France, 1990

F

Portuguese

Left arm

32

32

33

Prednisolone 0.5 mg/kg/d

Fever, arthralgia

Absence of left radial pulse

Supra-aortic arteries, abdominal aorta

 

Perniciaro et al. [17], USA, 1987

M

 

Left popliteal area

17.5

19

19

Systemic corticosteroids

Intermittent fever

   

Case 1

F

Chinese

Bilateral thighs and buttocks, upper extremities

24

24

28

Prednisolone 40 mg/d, cyclosporine 0.1 g/d, triptolide 60 mg/d

Fever

Dizziness, pulseless in both upper limbs

The right innominate artery (subclavian artery, common carotid artery) was completely obliterated, and the left common carotid artery was entirely occluded. The initial segment of the left vertebral artery had stenosis, and the left vertebral artery was enlarged

 

Case 2

F

Chinese

Both lower and upper extremities, left buccal region, face

No record

17

24

Methylprednisolone

 

Left arm weakness and dizziness

The left carotid artery, left subclavian artery and bilateral femoral arteries, the superficial femoral arteries and left popliteal artery, the ascending aorta,aortic arch and its branches, descending aorta, abdominal aorta. The celiac stem and mesenteric artery were occluded, with multiple collateral circulation formation. The main hepatic artery and splenic artery

 
  1. PG pyoderma gangrenosum, TA Takayasu arteritis, RP relapsing polychondritis