scholarly journals 17. Angiolymphoid hyperplasia with eosinophilia mimicking temporal arteritis

2019 ◽  
Vol 3 (Supplement_1) ◽  
Author(s):  
Ghazal Ansari ◽  
Angela Pakozdi ◽  
Oseme Etomi

Abstract Introduction A 32-year-old man presented to rheumatology clinic with a 2-year history of progressively worsening right temporal headache, a painful hard lump on his right temple and pre-auricular tenderness not responding to conventional analgesics. Patient denied any jaw and tongue claudication. His vision was not compromised and patient denied weight loss, night sweats and arthralgia. He was fit and well otherwise. Case description Examination revealed a pulsatile mass with a length of 1 cm along the superficial temporal artery. Temporal artery ultrasound revealed lump corresponds to the superficial artery with thickened wall and halo formation raising the possibility of temporal arteritis. MRI head scan suggested inflammation of the superficial temporal artery. Discussion Because of the atypical features of young age and normal inflammatory markers, corticosteroid treatment was not commenced. A temporal artery biopsy showed no evidence of vasculitis, but revealed reactive lymphoid follicles with eosinophil infiltrates in the surrounding soft tissue. The diagnosis of angiolymphoid hyperplasia with eosinophilia (ALHE) was made. The patient’s headaches have completely resolved after the temporal biopsy. Unfortunately, three months later the lump and the headaches recurred. The symptoms improved with topical tacrolimus 0.1% ointment and he underwent for complete surgical excision of the lump. Patient is asymptomatic and remained well. Key learning points We present a case of angiolymphoid hyperplasia with eosinophilia which can mimic temporal arteritis presentation as in our case and has totally different approach to management.  ALHE, which is a benign, locally proliferative condition with predilection to the periauricular and scalp area with no definitive treatment however, complete excision can be curative. It may recur in up to one-third of cases in the absence of complete surgical excision as in our patient. Conflict of interest The authors declare no conflicts of interest.

2015 ◽  
Vol 139 (5) ◽  
pp. 683-686 ◽  
Author(s):  
Ruifeng Guo ◽  
Alde Carlo P. Gavino

Angiolymphoid hyperplasia with eosinophilia (ALHE), also named epithelioid hemangioma (EH), is an inflamed vascular tumefaction of uncertain pathogenesis, characterized by proliferation of histiocytoid endothelial cells with prominent lymphocytic and eosinophilic infiltration. Although considered a benign condition, it may recur in up to one-third of cases in the absence of complete surgical excision. The pathogenesis of ALHE/EH is still controversial. However, reaction to trauma and arteriovenous shunting are considered relevant. Histologically, ALHE/EH may be differentiated from other vascular neoplasms by its several unique characteristics including prominent proliferation of plump endothelial cells, and accompanying eosinophilic and lymphocytic inflammation, often with formation of lymphoid follicles. Surgery is the mainstay of treatment and various other treatment strategies have been used with varying results.


Rheumatology ◽  
2021 ◽  
Vol 60 (Supplement_1) ◽  
Author(s):  
Jessica Ellis ◽  
Keziah Austin ◽  
Sarah Emerson

Abstract Background/Aims  A 49-year-old female of Nepalese heritage was referred with right-sided headache, scalp tenderness, and a painful swelling overlying the right temple. She denied any visual or claudicant symptoms but felt systemically unwell with a fever. There were no symptoms suggestive of an inflammatory arthritis, underlying connective tissue disease or vasculitis. She was normally fit and well with no past medical history. She did not take any regular medications and denied using over the counter or illicit drugs or recent travel. On review she had a low grade fever. There was a large tender, erythematous swelling overlying the right temple. Bilaterally the temporal arteries were palpable and pulsatile. Peripheral pulses were normal with no bruits. There was no evidence of shingles (HSV) or local infection. Full systemic examination revealed no other abnormalities. Laboratory tests showed: PV 2.56, CRP 101, total white cell count 14.38 (eosinophils 0.4), albumin 33, Hb 115. Urine dip was normal. Renal function, liver function and immunoglobulins were normal. ANCA was negative. Hypoechogenicity surrounding the right frontal branch of the right temporal artery was seen on ultrasound. There were no discrete masses suggestive of cysts, abscess or tumours. Temporal artery biopsy confirmed the presence of vasculitis; histology demonstrated transmural lymphohistiocytic inflammation, disruption of the elastic lamina and intimal proliferation. Prednisolone was started at 40mg daily. Four weeks after initially presenting she was asymptomatic and her inflammatory markers had normalised. Methods  The case is discussed below. Results  Temporal arteritis, or GCA, is primarily a disease of older adults; with age 50 often used as an inclusion criteria, and is more common in Caucasian populations. Limited reports exist of GCA in younger cohorts, but these are rare. An important differential in younger patients, such as ours, is juvenile temporal arteritis. This rare localised vasculitis affects almost exclusively the temporal artery. It is typically a disease of young males, who present with non-tender temporal swelling. Systemic symptoms are unusual and inflammatory markers are normal. Clinical or laboratory evidence of organ involvement, peripheral eosinophilia or fibrinoid necrosis on histology should prompt consideration of an AAV or PAN. Incidence of GCA increases in correlation with Northern latitude, with highest rates reported in Scandinavian and North American populations. GCA is rare in Asian populations. Higher diagnostic rates in countries where physicians have increased awareness of GCA proposed as an explanation for this difference; however differences in incidence are still observed between Asian and Caucasian populations presenting to the same healthcare providers. Conclusion  GCA is an uncommon diagnosis in younger and non-Caucasian patients. Thorough investigation through ultrasound and biopsy helped increase our diagnostic confidence in this unusual case. Rheumatologists must be alert to atypical presentations in order to deliver prompt and potentially sight-saving treatment. Disclosure  J. Ellis: None. K. Austin: None. S. Emerson: None.


2015 ◽  
Vol 209 (2) ◽  
pp. 338-341 ◽  
Author(s):  
Khoi Le ◽  
Lindsay M. Bools ◽  
Allison B. Lynn ◽  
Thomas V. Clancy ◽  
W. Borden Hooks ◽  
...  

Author(s):  
Jan Tore Gran

Polymyalgia rheumatica and temporal arteritis are distinct but overlapping inflammatory conditions of unknown aetiology. They almost exclusively affect people over 50 years of age, women more than men (ratio 2–3:1), and particularly those of Nordic heritage. Temporal arteritis is characterized by granulomatous inflammation that penetrates all layers of the wall of medium and (often) large muscular arteries, in particular the superficial temporal artery. Histological examination of tissues from patients with polymyalgia rheumatica shows nonspecific changes only. The term ‘giant cell arteritis’ is properly used only to describe patients with biopsy-proven arteritis....


1970 ◽  
Vol 20 (1) ◽  
pp. 78-81
Author(s):  
NK Sinha ◽  
MH Rashid ◽  
MM Shaheen ◽  
DC Talukder ◽  
MAY Fakir ◽  
...  

Juvenile angiofibroma is a rare hypervascular, locally aggressive benign tumour which is exclusively found in the nose and paranasal sinuses of male adolescents. The definitive treatment for this tumour is complete surgical excision. Different surgical approaches are used for complete excision. Most recent development is excision of the tumour using endoscopes. But in certain cases with large size and different extensions, open transfacial approaches are the choice for complete removal and for less operative bleeding, which are the main challenges for surgical excision of this tumour. DOI: http://dx.doi.org/10.3329/jdmc.v20i1.8587 J Dhaka Med Coll. 2011; 20(1) :78-81


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