Elsevier

The Journal of Pediatrics

Volume 145, Issue 6, December 2004, Pages 826-831
The Journal of Pediatrics

Original Article
Pulmonary arteriovenous malformations in children: Outcomes of transcatheter embolotherapy

https://doi.org/10.1016/j.jpeds.2004.08.046Get rights and content

Objective

To describe outcomes of transcatheter embolotherapy (TCE) in children with pulmonary arteriovenous malformations (PAVMs).

Study design

Chart and imaging review of all children (age ≤18 years) treated for PAVMs by TCE at three hereditary hemorrhagic telangiectasia centers.

Results

All 42 treated patients were included, with a mean age of 12 years (range, 4 to 18). Cyanosis was present in 25 of 42 patients (60%). Hemoptysis had occurred in 3 of 42 patients (7%) and neurologic complications (stroke, cerebral abscess) occurred in 8 patients (19%) before assessment. PAVMs were focal in 30 of 42 (71%) and diffuse in 12 of 42 (29%) patients. TCE was performed for 172 PAVMs and 35 diffuse regions (regional TCE). Follow-up was obtained in 38 of 42 (90%) patients (mean, 7 years). After TCE in patients with focal PAVMs, oxygenation improved significantly, with no further complications from the PAVMs. Reperfusion was noted in 23 of 153 (15%) PAVMs. Eighteen of 23 (78 %) of these were retreated, with documented aneurysmal involution in 10 of 13 (77%) patients. TCE complications included pleuritic chest pain (24% of sessions) and deployment complications (device paradoxical embolization or device misplacement) (3% of sessions, 1% of PAVMs), with no long-term complications.

Conclusions

PAVMs cause life-threatening complications in children; treatment with TCE is safe, with complication rates comparable to adult rates.

Section snippets

Subject selection

From the databases of 617 consecutive patients with PAVMs treated at Yale University (1988 to 2002), Johns Hopkins (1978 to 1988), and University of Toronto (1990 to 2002) HHT Centers, all 42 subjects ≤18 years of age who had undergone TCE for PAVMs were selected. Four included patients (diffuse patients 7, 8, 10, and 12) have been previously reported.13 Patients were referred to the HHT Centers either for suspected PAVMs or screening in asymptomatic children of HHT families, and investigators

Baseline characteristics and clinical presentation

All 42 patients meeting inclusion criteria were included. PAVMs were focal in 30 of 42 (71%) patients and diffuse in 12 of 42 (29%); 24 of 42 (57%) reported dyspnea on exertion or exercise intolerance (Table I). Three of 42 (7%) reported previous hemoptysis (minor), and none had previous spontaneous hemothorax. Cyanosis was present in 25 of 42 (60%) and clubbing was present in 19 of 42 (45%). Neurologic complications of PAVMs (transient ischemic attack, MRI-detected stroke, and cerebral

Discussion

PAVMs have been well described in adults, but there is a paucity of literature regarding the presentation and management of PAVMs in children. We report 42 children with PAVMs, the largest series in the literature to date, describing the safety and efficacy of TCE. We have demonstrated that children with PAVMs can have life-threatening complications. Yet, these patients can be treated safely and efficaciously with TCE.

PAVMs can lead to symptoms and complications in children. More than half of

References (16)

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Supported by the Squires Club, Nelson Arthur Hyland Foundation, Sonor Foundation, St Michael's Hospital Research Institute (M.E.F.), and March of Dimes grant HHT-FY03-677, Josephine Lawrence Hopkins Foundation, and General Clinical Research Center NIH grant M01-RR-00125 (R.I.W., K.H., and A.T.).

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