An unusual case of lower limb dysplasia with ulcer after interventional closure of PDA in childhood

doi:10.21203/rs.3.rs-2327625/v1

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Case Report

An unusual case of lower limb dysplasia with ulcer after interventional closure of PDA in childhood

https://doi.org/10.21203/rs.3.rs-2327625/v1

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Background: Lower limb dysplasia is a rare complication after interventional closure of patent ductus arteriosus (PDA) in childhood. The most common treatment of PDA is interventional closure via femoral artery. Compared with traditional surgery, its advantages include less trauma and faster postoperative recovery, but the complications after interventional occlusion should not be ignored.

Case presentation: A 10-year-old girl had redness and swelling of the toes of the right lower limb with skin ulceration on the medial side of the right foot for more than 4 months. She accepted interventional closure of PDA 7 years ago. Physical examination showed that the right foot was about 2 cm shorter than the left foot. We present a rare case of a young girl with lower limb dysplasia after interventional closure of PDA in childhood, Combined findings of Color Doppler ultrasound, Intravenous Ultrasound (IVUS), Computed tomography angiography (CTA) and Angiography, we found right foot developmental delay most likely was due to vascular injury after interventional closure of PDA in childhood.

Conclusion: It is critical to understand not only the acute but also the long-term complications that may occur after interventional procedure. To avoid the development of vascular injury, it is advised that patients who underwent femoral artery catheterization for interventional treatment in their early years undergo strict clinical and imaging surveillance.

Lower limb dysplasia

Interventional treatment

vascular injury

Patent ductus arteriosus

Children

Intravenous Ultrasound

Long-term complications

The ductus arteriosus (DA) is a fetal vascular connection between the main pulmonary artery and the aorta that diverts blood away from the pulmonary bed. After birth, the DA undergoes active constriction and eventual obliteration. A PDA occurs when the DA fails to completely close postnatal ^[1]. The clinical manifestation of PDA is determined by the degree of left-to-right shunt, which depends on the size and length of the PDA, as well as pulmonary and systemic vascular resistance. Transfemoral artery interventional closure is the most common method for the treatment of PDA. Compared with traditional surgery, its advantages include less trauma and faster postoperative recovery, but the complications after interventional occlusion should not be ignored. Catheter-related arterial thrombosis (CAT) is the damage to the vascular wall after the catheter is inserted into the artery, leading to the formation of intravascular thrombus ^[2].

We present a rare case of a young girl with longterm complications after PDA interventional closure.

A 10-year-old girl was admitted to the hospital due to "redness and swelling of the toes of the right lower limb with skin ulceration on the medial side of the right foot for more than 4 months". Physical examination showed that the right foot was about 2 cm shorter than the left foot, the first and second toes of the right foot were red and swollen, ulceration was seen on the medial side of the right foot, the surrounding skin was dry, increased capillary shadows were observed on the skin surface of the medial segment of the right foot, and the right dorsalis pedis artery pulse was weakened. Medical history: underwent interventional closure of PDA via right femoral artery 7 years ago and the pulse of patient’s right dorsalis pedis artery was weakened.

The erythrocyte sedimentation rate was slightly elevated after admission, but the antinuclear antibodies, vascular inflammatory factors, immune-related protein antibodies and BCR-ABL gene test findings were negative, and the coagulation factor results were within the reference value range. Heterogeneous stenosis of the right artery below the thoracic aortic plane, decreased resistance of arterial blood flow in the right lower limb, and subcutaneous lymphadenectasis in the right inguinal region were found by Color Doppler Ultrasound. CTA of the celiac artery and lower limb arteries revealed stenosis at the origin of the celiac trunk, with a narrowest diameter about 0.2 cm, as well as the segmental mild stenosis of the right femoral artery and popliteal artery, and magnetic resonance imaging (MRI) of the right foot revealed a minimal bone marrow edema at the distal end of the first and second phalanges of the right foot and the middle segment of the right tibia. Lower limb arteriography was performed to help clarify the findings.

Angiography revealed that the branches of the right deep femoral artery and the upper segment of the superficial femoral artery were rough, indicating local vascular segmental stenosis or even occlusion; scattered filling defects could be seen in the right popliteal artery, indicating plaque formation with mild stenosis; the main trunk of the right anterior tibial artery and the peroneal artery showed beaded changes (Fig. 1), indicating occlusion, and the blood was supplied from the collateral artery to the dorsalis pedis artery; the dorsalis pedis artery was poorly visualized, disorganized anastomotic branches could be seen. The results of intraoperative inguinal lymph node puncture demonstrated lymphoid tissue hyperplasia, sinus opening, and visible lymphoid follicles, indicating reactive hyperplasia.

After the patient was admitted to the hospital, she was treated with intravenous prostaglandin, and felt the pain was relieved. Then balloon dilatation angioplasty of the right popliteal and anterior tibial arteries was performed under local anesthesia, at the same time, IVUS was used to check the tunica intima during operaion and revealed that the right femoral artery collapsed with thrombosis and the intimal of the artery was negative vascular remodeling and less smooth (Fig. 2). The patient's general condition was good during the operation, but after the operation, he developed a large skin rash with mild pruritus and no other symptoms such as dyspnea and chest pain. Considering allergic reaction to the contrast agent, it improved significantly after symptomatic treatment. One day after the operation, the patient reported that the pain in the toes of the lower limbs was significantly improved, and the redness and swelling of the first toe of the right lower limb was significantly improved on examination.

Femoral artery thrombosis was relatively uncommon during the catheterization of femoral artery for interventional treatment in children, but if it is not treated properly, it will cause serious consequences, including the development and dysfunction of the affected limb. During left cardiac catheterization or interventional therapy via femoral artery, the incidence of femoral artery thrombosis remained 0.8 ~ 3.4 percent ^[3]. It is generally believed that the diameter of the catheter should not exceed 1/3 of the inner diameter of the artery ^[4]. When the catheter’s diameter exceeds 1/2 of the blood vessel’s diameter, it has a significant impact on hemodynamics and causes blood stasis in this area; additionally, the larger the diameter, the easier it is to rub the vascular endothelium, resulting in mechanical damage and increasing the incidence rate of thrombosis and thromboangiitis.

With the patient’s younger age and longer intubation time, the risk of arterial spasm increases and transient arterial obstruction may result in weakened arterial pulsation, whitening, pain and rarely skin necrosis ^[5]. When a patient’s distal circulation is impeded, such as a weakened or absent pulse, white or mottled skin, and cold distal limbs, arterial thrombosis and embolism should be suspected. In this case, the patient’s right lower limb toes were red and swollen, with skin ulceration on the medial side of the foot and diminished pulsation of the right dorsalis pedis artery, which was consistent with the clinical manifestations of redness and swelling of the right toe with skin ulceration caused by distal artery occlusion. The erythrocyte sedimentation rate of this patient was slightly higher, which was considered to be the vascular inflammatory change in acute stage, when combined with the pathological results of lymph node biopsy. According to reports, a retrospective study of 300 patients after cardiac catheterization in a literature article found that 7 pediatric patients, with an average age of 10 years, had an average ipsilateral femoral shortening of 2 cm after interventional therapy, and the length difference may be caused by vascular wall injury during the treatment of patients with congenital heart disease via the femoral artery pathway, and long-term vasospasm or thrombotic occlusion of the nutrient artery after operation^[6]. In this case, the pediatric patient’s lower limbs were of identical length, but the right foot was about 2 cm shorter than the left foot, and PDA interventional closure was performed via right femoral artery 7 years ago. Color Doppler ultrasound revealed the blood flow resistance index of right lower limb artery reduced, CTA revealed stenosis of the initial segment of the celiac trunk and arteriography showed occlusion of the right anterior tibial artery, peroneal artery and dorsalis pedis artery. The cause of right foot shortening was considered to be a growth and development disorder of the right foot caused by vascular stenosis and long-term occlusion of right lower limb.

For the diagnosis and treatment of congenital heart disease, the investigations or treatments via vascular intervention in neonates and children are critical. Studies have shown that the detection rate of arterial thrombosis in children increased from 8.3 percent to 23.4 percent when conventional Color Doppler Ultrasonography was performed after interventional therapy, and in multivariate analysis, lower body weight, larger arterial sheath, and longer operation time were independently associated with a higher chance of thrombosis. It is critical to assess the state of limb pulsation after interventional procedure ^[7]. If the pulse becomes weak or vanishes, release the bandage immediately, constantly monitor the patient, strengthen anticoagulant therapy and regularly monitor the patient to avoid skeletal abnormalities. Macnicol believes that distal thrombosis may contribute to local persistent ischemia of the growth plate ^[8]. It is critical to understand not only the acute but also the long-term complications that may occur.

In conclusion, despite the low prevalence of vascular etiology in bone growth length disparities, the importance of informing parents and pediatricians about these potential complications is highlighted based on the findings of related studies ^[9]. To avoid the development of vascular injury, it is advised that patients who underwent femoral artery catheterization for interventional treatment in their early years undergo strict clinical and imaging surveillance.

PDA

patent ductus arteriosus

CTA

Computed tomography angiography

IVUS

Intravenous Ultrasound

ductus arteriosus

CAT

Catheter-related arterial thrombosis

MRI

magnetic resonance imaging.

Ethics approval and consent to participate

Not applicable.

Consent for publication

Written consent to publish this information was obtained from the parents of the study participant. Proof of consent to publish from study participant can be requested at any time.

Availability of data and materials

Data on patient and case details are available from the authors on reasonable request.

Competing interests

The authors declare that they have no competing interests.

Funding

This research was funded by a grant (No. 2021-201) from the Health Commission of Sichuan Province. Yan Deng the corresponding author was supported by the fund, and the Health Commission of Sichuan Province provided support to collect data.

Authors’ contributions

ZL contributed to manuscript drafting and reviewed the literature; YD was responsible for revising the manuscript for important intellectual content; PX and JL provide the pictures; QX and RY reviewed the literature. all authors issued the final approval for the version to be submitted.

Acknowledgements

We would like to thank the patient and her family for their consent to publish this report. We appreciate the doctors' cooperation and participation in this study.

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Crameri O, et al. Treatment of Catheter-Related Arterial Thrombosis in Children: A 15-Year Single- Center Experience. J Pediatr. 2021;239:182–6.
Rizzi M, et al. Catheter-Related Arterial Thrombosis in Neonates and Children: A Systematic Review. Thromb Haemost. 2018;118(6):1058–66.
Barone G, et al. A Systematic Ultrasound Evaluation of the Diameter of Deep Veins in the Newborn: Results and Implications for Clinical Practice. Neonatology. 2019;115(4):335–40.
Feltes TF, et al. Indications for cardiac catheterization and intervention in pediatric cardiac disease: a scientific statement from the American Heart Association. Circulation. 2011;123(22):2607–52.
Díaz-Ben B, et al. Discrepancia de longitud de los miembros inferiores tras canalización arterial en niños [LIMB LENGH SHORTENING AFTER ARTERIAL CANNULATION IN INFANCY]. Rev Esp Cir Ortop Traumatol. 2016;60(1):12–9.
Kamyszek RW, et al. Routine postprocedure ultrasound increases rate of detection of femoral arterial thrombosis in infants after cardiac catheterization. Catheter Cardiovasc Interv. 2019;93(4):652–9.
Macnicol MF. Arrest of the growth plate after arterial cannulation in infancy. J Bone Joint Surg Br. 2000;82(2):172–5.
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No competing interests reported.

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An unusual case of lower limb dysplasia with ulcer after interventional closure of PDA in childhood

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