Cyanosis for more than 4 years in a girl aged 4 years and 3 months

WU Mao-Lan; ZHENG Xiang-Rong; ZHU Yan-Tong; ZHANG Guo-Yuan; LIU Chen-Tao; WANG Xia; TANG Yong-Jun

doi:10.7499/j.issn.1008-8830.1910147

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Chinese Journal of Contemporary Pediatrics ›› 2020, Vol. 22 ›› Issue (4) : 387-390. DOI: 10.7499/j.issn.1008-8830.1910147

CASE ANALYSIS

Cyanosis for more than 4 years in a girl aged 4 years and 3 months

WU Mao-Lan, ZHENG Xiang-Rong, ZHU Yan-Tong, ZHANG Guo-Yuan, LIU Chen-Tao, WANG Xia, TANG Yong-Jun

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Abstract

A girl, aged 4 years and 3 months, presented with cyanosis of the lips shortly after birth. She then experienced shortness of breath after activity 1 year ago and acrocyanosis 3 months ago, with obvious acropachy and toe deformity. Laboratory examinations revealed an increase in hemoglobin (178 g/L) and a reduction in arterial partial pressure of oxygen (37.7 mm Hg). Plain and contrast-enhanced CT scans of the lungs showed a large area of dense shadow and multiple nodules with clear boundaries in the right lower lung, as well as thickening of the arteries and dilatation of the veins in the right lower lung. Magnetic resonance angiography of the pulmonary artery showed large arteriovenous malformation in the lung. The child was diagnosed with congenital pulmonary arteriovenous fistula and was given interventional embolization of the pulmonary arterial fistula. The child was followed up at 3 months after surgery. The symptoms of shortness of breath and cyanosis disappeared, and activity tolerance, heart rate, hemoglobin, red blood cell count, and transcutaneous oxygen saturation all returned to normal.

Key words

Pulmonary arteriovenous fistula / Cyanosis / Interventional embolization therapy / Child

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WU Mao-Lan, ZHENG Xiang-Rong, ZHU Yan-Tong, ZHANG Guo-Yuan, LIU Chen-Tao, WANG Xia, TANG Yong-Jun. Cyanosis for more than 4 years in a girl aged 4 years and 3 months[J]. Chinese Journal of Contemporary Pediatrics. 2020, 22(4): 387-390 https://doi.org/10.7499/j.issn.1008-8830.1910147

References

[1] Shovlin CL, Condliffe R, Donaldson JW, et al. British Thoracic Society clinical statement on pulmonary arteriovenous malformations[J]. Thorax, 2017, 72(12):1154-1163.
[2] Ghelani SJ, Rathod RH. Pulmonary arteriovenous malformations:the consequences of bypassing the capillary bed[J]. J Thorac Cardiovasc Surg, 2015, 150(3):717-719.
[3] Tellapuri S, Park HS, Kalva SP. Pulmonary arteriovenous malformations[J]. Int J Cardiovasc Imaging, 2019, 35(8):1421-1428.
[4] Hsu CC, Kwan GN, Evans-Barns H, et al. Embolisation for pulmonary arteriovenous malformation[J]. Cochrane Database Syst Rev, 2018, 1:CD008017.
[5] Nakayama M, Nawa T, Chonan T, et al. Prevalence of pulmonary arteriovenous malformations as estimated by lowdose thoracic CT screening[J]. Intern Med, 2012, 51(13):1677-1681.
[6] 吴琳, 盛锋, 刘芳, 等. Amplatzer血管塞子堵闭儿童先天性肺动静脉瘘[J]. 中国介入影像与治疗学, 2007, 4(6):486-487.
[7] 张志芳, 陈轶维, 李奋, 等. 先天性儿童肺动静脉瘘介入封堵治疗效果分析[J/CD]. 中华临床医师杂志(电子版), 2012, 6(22):7439-7441.
[8] 谢兆丰, 张智伟, 李渝芬, 等. 儿童期肺动静脉瘘9例报告[J]. 临床儿科杂志, 2013, 31(5):491-492.
[9] 周雪, 高伟, 易岂建, 等. 经导管封堵儿童先天性肺动静脉瘘的临床报告[J]. 重庆医科大学学报, 2018, 43(3):357-361.
[10] Pollak JS, Saluja S, Thabet A, et al. Clinical and anatomic outcomes after embolotherapy of pulmonary arteriovenous malformations[J]. J Vasc Interv Radiol, 2006, 17(1):35-44.
[11] Faughnan ME, Thabet A, Mei-Zahav M, et al. Pulmonary arteriovenous malformations in children:outcomes of transcatheter embolotherapy[J]. J Pediatr, 2004, 145(6):826-831.
[12] 陈和斌, 陆小霞, 蒋鲲. 儿童反复咯血的病因及临床诊治分析[J]. 中国当代儿科杂志, 2014, 16(3):281-284.
[13] Shovlin CL, Letarte M. Hereditary haemorrhagic telangiectasia and pulmonary arteriovenous malformations:issues in clinical management and review of pathogenic mechanisms[J]. Thorax, 1999, 54(8):714-729.
[14] Santhirapala V, Williams LC, Tighe HC, et al. Arterial oxygen content is precisely maintained by graded erythrocytotic responses in settings of high/normal serum iron levels, and predicts exercise capacity:an observational study of hypoxaemic patients with pulmonary arteriovenous malformations[J]. PLoS One, 2014, 9(3):e90777.
[15] Vorselaars VMM, Hosman AE, Westermann CJJ, et al. Pulmonary arterial hypertension and hereditary haemorrhagic telangiectasia[J]. Int J Mol Sci, 2018, 19(10). pii:E3203.