scholarly journals Giant bilateral axillary artery aneurysms with left complete obstructive thrombus in intravenous immunoglobulin-sensitive Kawasaki disease: a case report

2021 ◽  
Vol 19 (1) ◽  
Author(s):  
Chen Chu ◽  
Lan He ◽  
Yi-xiang Lin ◽  
Li-ping Xie ◽  
Fang Liu

Abstract Background Kawasaki disease (KD) is a systemic vasculitis that predominantly affects medium-sized arteries. In addition to well-known coronary artery aneurysms (CAAs), peripheral systemic artery aneurysms (SAAs) have also been sporadically reported. In the literatures, SAAs occurred mainly in untreated, intravenous immunoglobin (IVIG)-resistant, or severe refractory KD, and thrombotic events in SAAs were rarely reported. Case presentation A 10-month-old boy with a history of KD was referred to our hospital for suspected pseudoaneurysm of the axillary arteries. Four months prior to presentation, he had persistent fever, conjunctival congestion, and rash. On the 10th day of fever echocardiogram showed biliteral CAAs. He was then diagnosed with KD and given IVIG 2 g/kg and aspirin at a local hospital. His fever and symptoms soon subsided and he was discharged with low dose aspirin and dipyridamole. One month prior to presentation, his parents incidentally palpated swellings in his bilateral axillae. On admission, physical examination revealed a pulsatile swelling in his right axilla and a non-pulsatile swelling in the left with impalpable left brachial and radial pulses, cooler and less active left upper limb than the right one. While the pulses of other three limbs were normal. Ultrasound examination revealed giant bilateral axillary artery aneurysms (AAAs) with massive thrombus in the left. Angiography confirmed giant bilateral AAAs with left AAAs completely occluded and fine collateral vessels connecting to the distal brachial artery, in addition to giant bilateral multiple CAAs without stenoses. The patient was given intravenous prostaglandin for 10 days to allow for formation of collateral circulation, as well as aspirin, low molecular weight heparin (which was switched to warfarin before discharge) and metoprolol. At discharge, the temperature and movement of his left upper limb improved significantly. On follow-up at 7 months, his left upper limb further improved and was similar to the right with no occurrence of cardiovascular events. The images of CAAs and AAAs on echocardiogram and computerized tomography remained the same. Conclusions This case highlights the importance of evaluating peripheral SAAs in KD patients with CAAs, even if their course of treatment appears smooth. For both large non-aortic SAAs and CAAs in KD patients, antithrombotic therapy is of utmost importance.

2021 ◽  
Author(s):  
Chen Chu ◽  
Lan He ◽  
Yi-xiang Lin ◽  
Fang Liu

Abstract Background: Kawasaki disease (KD) is a systemic vasculitis that predominantly affects medium-sized arteries. In addition to well-known coronary artery aneurysms (CAAs), peripheral systemic artery aneurysms (SAAs) have also been sporadically reported. In the literatures, SAAs occurred mainly in untreated, intravenous immunoglobin (IVIG)-resistant, or severe refractory KD, and thrombotic events in SAA were rarely reported.Case presentation: A 10-month-old boy with history of KD was referred to our hospital for suspected pseudoaneurysm of the axillary arteries. Four months prior to presentation, he had recurrent fever, conjunctival congestion, and rash. On the 10th day of fever echocardiography showed biliteral CAAs. He was then diagnosed with KD and given IVIG 2g/kg and aspirin at a local hospital. His fever and symptoms soon subsided and he was discharged with low dose aspirin and dipyridamole. One month prior to presentation, his parents incidentally palpated swellings in his axillae bilaterally. On admission, physical examination revealed a pulsatile swelling in his right axilla and a non-pulsatile swelling in the left. His left brachial and radial pulses were not palpable, while the pulses of other three limbs were normal. His left upper limb was cooler and less active compared to the right. Ultrasound examination revealed bilateral giant axillary artery aneurysms (AAAs) with massive thrombus in the left. Angiography confirmed bilateral giant AAAs with left AAAs completely embolized and fine collateral vessels connecting to the distal brachial artery, in addition to bilateral multiple giant CAAs without stenoses. The patient was given intravenous prostaglandin for ten days to allow for formation of collateral circulation, as well as aspirin, low molecular weight heparin (which was switched to warfarin before discharge) and metoprolol. At discharge, the temperature and movement of his left upper limb improved significantly. On follow-up at three months, his left limb improved and was similar to the right with no cardiovascular event having occurred. The images of CAAs and AAAs on ultrasound and computerized tomography remained the same.Conclusions: This case highlights the importance of evaluating peripheral SAAs in KD patients with CAA, even if their clinical course appears uncomplicated. For both large non-aortic SAAs and CAAs in KD patients, antithrombotic therapy is of utmost importance.


2020 ◽  
Vol 15 (1) ◽  
pp. 110-113
Author(s):  
Md Abdur Razzak ◽  
Ghulam Kawnayn ◽  
Fateha Naznin ◽  
Quazi Audry Arafat Rahman

Moyamoya disease is a disease in which certain arteries in the brain are constricted. Blood flow is blocked by the constriction, and also by blood clots (thrombosis). A collateral circulation develops around the blocked vessels to compensate for the blockage, but the collateral vessels are small, weak, and prone to bleeding, aneurysm and thrombosis which may result in TIA, recurrent ischemic or hemorrhagic stroke or seizure. The disease may manifest in pediatric age or young adults. In May 2019 we have diagnosed a young lady with Moyamoya disease who presented with right sided hemiplegia, motor aphasia and dysphagia. She was labeled as hypertensive 6 months prior to this event and used to take anti-hypertensive irregularly and gave past history of occasional headache. Her CT scan and MRI of brain revealed left sided ischemic infarct involving frontotemporoparietal region and cerebral angiogram revealed narrowing of left MCA and non-visualization of distal part. There is extensive fine collaterals (Moyamoya vessels) giving the appearance of puffed smoke. The right ACA and MCA were also narrowed with appearance of early collateral vessels. She was treated with aspirin, PPI, NG feeding, antihypertensive medication, physiotherapy, rehabilitation therapy and other supportive care. His condition gradually improved and discharged on 2.7.19. He was referred to Department of Neurosurgery for cerebral revascularization by STA-MCA (superficial temporal and middle cerebral arteries) bypass surgery after stabilization and MR perfusion study. Journal of Armed Forces Medical College Bangladesh Vol.15 (1) 2019: 110-113


2014 ◽  
Vol 10 (4) ◽  
pp. 91-94
Author(s):  
A Bhatnagar ◽  
M Deshpande

Servelle Martorelle Syndrome is a congenital vascular malformation associated with soft tissue hypertrophy and bony hypoplasia. This rarely involves whole of an extremity, with involvement of part of limbs reported in literature. We present a case of a twelve year boy who presented to the Department of Plastic Surgery SGPGIMS in April 2011 ,with history of circumferential soft tissue hypertrophy involving whole of left upper limb, scapular region and axilla since birth. The entire left upper limb length was lesser than the right upper limb. Hence this is a very rare case of Servelle Martorelle Syndrome having extensive limb involvement at a very young age. Highlighted is the role of conservative treatment and close follow-up to understand the natural history of the diseases, with prompt treatment of complications. DOI: http://dx.doi.org/10.3126/kumj.v10i4.11011 Kathmandu Univ Med J 2012;10(4):91-94


2014 ◽  
Vol 03 (04) ◽  
pp. 225-228
Author(s):  
Sankaran PK ◽  
Gunapriya Raghunath ◽  
Sathyan R. ◽  

AbstractDuring routine dissection of upper limb, a variation in the origin of radial artery was observed in a female cadaver of about 60 years. The radial artery is usually a smaller terminal branch of the brachial artery in the forearm. On the right side of the cadaver the radial artery was found to originate from second part of the axillary artery, whereas on the left side the origin of radial artery was found to be normal. This variant of radial artery has been rarely documented in literature. Accurate anatomy of radial artery and its variations have definite diagnostic, interventional, and surgical significance.


2018 ◽  
Vol 2018 ◽  
pp. 1-3 ◽  
Author(s):  
Tracey Dyer ◽  
Paul Dancey ◽  
John Martin ◽  
Suryakant Shah

Kawasaki disease (KD) is an acute systemic vasculitis of childhood. The diagnosis can be made in a patient who presents with a prolonged high fever and meeting at least four of five criteria including polymorphous rash, mucosal changes, extremity changes (including swelling and/or palmar and plantar erythema), bilateral nonsuppurative conjunctivitis, and unilateral cervical lymphadenopathy. Atypical KD refers to patients who have not met the full criteria and in whom atypical features may be present. We discuss a case of a 6-year-old male who presented to the Emergency Department with torticollis. A series of investigations for elevated inflammatory markers revealed dilated coronary artery aneurysms on echocardiogram, and thus he was diagnosed with atypical KD. His only other criteria were bilateral nonsuppurative conjunctivitis and a prior brief febrile illness. He was treated with high-dose intravenous immune globulin (IVIG) and low-dose aspirin. Low-molecular-weight heparin and atenolol were added due to the presence of giant aneurysms.


Children ◽  
2018 ◽  
Vol 5 (11) ◽  
pp. 155 ◽  
Author(s):  
Nikita Goswami ◽  
Katherine Marzan ◽  
Elizabeth De Oliveira ◽  
Sharon Wagner-Lees ◽  
Jacqueline Szmuszkovicz

Kawasaki disease (KD) is a self-limited systemic vasculitis, most often occurring in children 1–5 years old. It has a 2% recurrence rate and is associated with coronary aneurysms (CA), which can develop within two weeks of onset. A 25% increased risk is noted in patients who are recalcitrant to treatment. We describe a patient with recurrence of KD three times, approximately four years apart. A 10-year-old female with two previous episodes of KD, at 11 months and five years of age), in which she met five out of five criteria for KD and had no coronary involvement, presented with 15 days of fever, conjunctivitis and mucocutaneous changes. Infectious work-up was negative, and she was diagnosed with incomplete KD meeting three out of five criteria. An echocardiogram (ECHO) on day 12 revealed dilation of the right coronary artery (RCA) and left coronary artery (LCA). Treatment with intravenous immunoglobulin (IVIG) and high-dose aspirin was started at an outside hospital. After transfer, serial ECHOs showed evolving coronary aneurysms, left anterior descending (LAD) z-score + 8.2 and RCA z-score + 4.0. She received 10 mg/kg infliximab (day 18) and began clopidogrel. A cardiac MRI (day 20) demonstrated progression of the LAD aneurysm, with a z-score + 13, and warfarin was started. To our knowledge, this is the first report of recurrent KD occurring three times at ~5 year intervals.


2020 ◽  
Vol 13 (3) ◽  
pp. 1082-1090
Author(s):  
Rosy Setiawati ◽  
Vivid Umi Varidha ◽  
Giuseppe Guglielmi ◽  
Filippo Del Grande

Traumatic lesions of the axillary artery itself are limited to 2.9–9% of major arterial injuries. Pseudoaneurysms represent a pulsating encapsulated hematoma in communication with the lumen of a ruptured vessel. Traumatic pseudoaneurysm of the axillary artery is a rare sequela of injury to the shoulder region. We describe a case of posttraumatic pseudoaneurysm involving the axillary artery, which was initially misdiagnosed as an aggressive soft tissue tumor. The man presented 10 years after an injury from a fall from a tree with a slowly growing mass in the right upper limb region and reduced range of movement. This is a neglected case with a history of traditional massage. The patient presented a pathologic fracture of the right proximal humerus and dislocation of the glenohumeral joint. At the beginning, it was suggested to be a primary soft tissue tumor, but after several examinations, including comparable X-ray, ultrasound, and histopathology, the results did not support a soft tissue tumor. Magnetic resonance imaging and computed tomography angiography (CTA) finally confirmed the finding of a pseudoaneurysm of the right axillary artery associated with a huge hematoma with different age of the bleeding product and granulation tissue. This case demonstrates the necessity of early diagnosis of axillary artery pseudoaneurysm to prevent complications after a history of trauma. CTA is a useful modality to evaluate vascular injury and provides valuable information.


2019 ◽  
Vol 47 (8) ◽  
pp. 4014-4018 ◽  
Author(s):  
Bo Zhang ◽  
Yunpeng Hao ◽  
Yanfeng Zhang ◽  
Nuo Yang ◽  
Hang Li ◽  
...  

Background Kawasaki disease (KD) is an acute multisystem vasculitic syndrome that predominantly affects infants and young children. Neurological complications are rare in patients with KD and the diagnosis is challenging. We report a case of KD that manifested as bilateral facial nerve palsy and meningitis. Case report A 6-month-old boy presented with a 10-day history of fever. Four days before admission, the patient developed a rash, conjunctival injection, perioral and perianal excoriation, and bilateral facial nerve palsy. Brain magnetic resonance imaging was normal. Echocardiography showed dilated coronary arteries and coronary artery aneurysms. A cerebrospinal fluid examination showed an elevated leukocyte count. A diagnosis of KD was made, and the patient was treated with gamma globulin and aspirin. The patient’s fever subsided on the following day and the right-sided facial nerve palsy was relieved 1 month later. An 18-month follow-up showed that the left-sided facial nerve palsy persisted and the patient’s condition remained stable. Conclusion KD manifesting as bilateral facial nerve palsy and meningitis is extremely rare. Clinicians should be aware of this condition, and early diagnosis and appropriate treatment should be emphasized.


Neurosurgery ◽  
2004 ◽  
Vol 54 (4) ◽  
pp. 1019-1024 ◽  
Author(s):  
M. Jafer Ali ◽  
Bernard R. Bendok ◽  
Christopher C. Getch ◽  
Numa R. Gottardi-Littell ◽  
Stefan Mindea ◽  
...  

Abstract OBJECTIVE AND IMPORTANCE Prevention of rebleeding is the most important aspect of the management of hemorrhagic moyamoya disease, because rebleeding causes significant morbidity and mortality. CLINICAL PRESENTATION A 26-year-old male patient with a history of moyamoya disease since the age of 3 years and multiple strokes was in a semicomatose state at presentation. He was found to have intraventricular and periventricular hemorrhages abutting the atrium of the right ventricle. His hospital course was complicated by a second hemorrhage. Both bleeding events were believed to be secondary to a ruptured right lateral posterior choroidal aneurysm. INTERVENTION The aneurysm was excised and revealed histopathology consistent with a true saccular aneurysm. Frameless stereotactic guidance was used during surgery to minimize damage to collateral vessels and to shorten the surgical corridor. CONCLUSION The management of hemorrhagic moyamoya disease should be modified based on the source of hemorrhage and its relation to a specifically located aneurysm. In the case of aneurysms arising from the choroidal artery, the general belief is that most of these represent pseudoaneurysms and have a tendency to regress spontaneously. Because of the rebleeding risk, we recommend early intervention in treating ruptured intracranial aneurysms using the least invasive surgical techniques.


2021 ◽  
Vol 14 (1) ◽  
pp. e240981
Author(s):  
Riju Ramachandran ◽  
Anoop Vasudevan Pillai ◽  
Suyambu Raja ◽  
Sailakshmi Sailesh

Novel COVID-19 continues to intrigue medical professionals with its varied presentations. Though it affects the respiratory tract primarily, thrombogenesis has been the Achilles’ heel. A 44-year-old man diagnosed with COVID-19 presented with upper limb pain at a local hospital and was found to have thrombosis of the right axillary artery. Despite a successful embolectomy at the local hospital, there was re-occlusion of the axillary artery and the limb became ischaemic. He was referred to our institution by which time the limb became gangrenous above the elbow and had to be amputated. Extensive sloughing of the nerves was also seen in the local area. Hypercoagulability presenting with various manifestations is common in COVID-19 and needs early anticoagulation. We present this asymptomatic patient who lost a limb to this COVID-19 sequelae.


Sign in / Sign up

Export Citation Format

Share Document