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Uncommon complication of pulmonary arterial hypertension treatment with the parenteral use of treprostinil

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C L I N I C A L V I G N E T T E Complication of the parenteral use of treprostinil 79 (Tricumed Medizintechnik GmbH, Kiel, Germa‑

ny) was implanted (implantation of this pump system was described elsewhere by Desole et al3).

Abdominal ultrasonography carried out 28 days after implantation, during the first pump refill, showed several compartments filled with fluid that precluded pump refill and required empty‑

ing with a puncture (a total of 230 ml of bloody fluid). After further 28 days, a hematoma around the pump was detected again and 80 ml of flu‑

id was evacuated.

During the next pump refill (84 days after im‑

plantation), it was impossible to pass the mem‑

brane. The fluoroscopic examination revealed a 180° pump rotation (Figure 1A): the membrane was oriented inwards, and the back of the pump, towards the abdominal skin. The patient com‑

plained of pain in the subclavian area at the site Pulmonary arterial hypertension (PAH) is char‑

acterized by remodeling of pulmonary arteri‑

oles, which leads to right ventricular failure.1 The treatment of PAH involves the use of pros‑

tacyclin analogues, eg, treprostinil,1,2 which may be administered either intravenously or subcutaneously.

A 28‑year ‑old woman diagnosed with idio‑

pathic PAH was treated with treprostinil, bosen‑

tan, and sildenafil. Treprostinil was adminis‑

tered subcutaneously, using a microinfusion pump, and the dose was gradually increased.

On the second day of therapy, the patient devel‑

oped persistent erythema (Supplementary mate‑

rial, Figure S1). The optimal dose of treprostinil was reached at 66 ng/kg/min, and then a deci‑

sion was made to switch from subcutaneous to intravenous drug infusion. A Lenus Pro pump

Correspondence to:

Małgorzata Zielska, MD,  Department of Cardiology,  Pomeranian Medical university,  ul. Powstańców Wielkopolskich 72,  70-111 Szczecin, Poland,  phone: +48 91 466 13 78,  email:malzielska@gmail.com Received: September 9, 2020.

Revision accepted:

October 25, 2020.

Published online:

November 4, 2020.

Kardiol Pol. 2021; 79 (1): 79-80 doi:10.33963/KP.15672 Copyright by the Author(s), 2021

C L I N I C A L V I G N E T T E

Uncommon complication of pulmonary arterial hypertension treatment

with the parenteral use of treprostinil

Małgorzata Zielska1, Maciej Lewandowski1, Katarzyna Widecka1, Marcin Kurzyna2, Piotr Kędzierski2, Naser Dib3, Jarosław Kaźmierczak1, Małgorzata Peregud ‑Pogorzelska1 1  Department of Cardiology, Pomeranian Medical university, Szczecin, Poland

2  Department of Pulmonary Circulation and Thromboembolic Diseases, Medical Centre of Postgraduate education, european Health Centre Otwock, Otwock, Poland 3  Department of Cancer Surgery, european Health Center Otwock, Otwock, Poland

Figure 1 A – fluoroscopic image of the rotated LenusPro pump; B – twisted catheter for intravenous infusion of treprostinil, found during a reoperation 84 days after pump implantation (arrow)

A B

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KARDIOLOGIA POLSKA 2021; 79 (1) 80

4 Kurzyna M, Małaczyńska -rajpold K, Koteja A, et al. An implantable pump  Lenus pro® in the treatment of pulmonary arterial hypertension with intravenous  treprostinil. BMC Pulm Med. 2017; 17: 162.

5 richter MJ, Harutyunova S, Bollmann T, et al. Long -term safety and outcome  of intravenous treprostinil via an implanted pump in pulmonary hypertension. 

J Heart Lung Transplant. 2018; 37: 1235-1244.

of catheter implantation, which persisted for a few days. The catheter position was normal on radiography and ultrasonography.

A revision surgery was performed to correct the position of the pump and catheter (several twists of the catheter were found, but these for‑

tunately did not cause occlusion) (Figure 1B). The re‑

vision was complicated by the formation of he‑

matoma, which required surgical intervention and drainage. No complications occurred dur‑

ing further 10‑month follow ‑up. Despite the pre‑

vious complications, the patient reported bet‑

ter quality of life. The clinical and biochemi‑

cal parameters of the patient improved over a 10‑month follow ‑up period (Supplementary material, Figure S2).

The continuous infusion of treprostinil is associated with potential complications. Pain at the injection site is the most commonly re‑

ported complication of using the subcutaneous administration route, which forces dose reduc‑

tion or drug withdrawal.1 The use of an intrave‑

nous pump eliminates discomfort and the risk of skin reaction at the injection site, which improves the health ‑related quality of life.4 Nevertheless, a therapy based on the use of an intravenous in‑

fusion pump may also be associated with com‑

plications. Richter et al5 reported that 60 out of 129 patients with an implanted Lenus Pro pump had complications. In a single case, the pump was displaced and required surgical repositioning. To our knowledge, this is the first report describing the case of pump rotation with multiple twists of the catheter causing discomfort due to pulling pain in the subclavian area yet no disturbance in drug delivery. Patients with symptoms (pain and discomfort) at the site of pump implantation or catheter entrance into the vein require specific diagnostic workup, including fluoroscopy, to as‑

sess the position of the pump.

SupplementAry mAteriAl

Supplementary material is available at www.mp.pl/kardiologiapolska.

Article informAtion

conflict of intereSt None declared.

open AcceSS This is an Open Access article distributed under the terms  of  the  Creative  Commons  Attribution -Non  Commercial -No  Derivatives  4.0  in- ternational License (CC BY -NC -ND 4.0), allowing third parties to download ar- ticles and share them with others, provided the original work is properly cited,  not changed in any way, distributed under the same license, and used for non- commercial purposes only. For commercial use, please contact the journal office  at kardiologiapolska@ptkardio.pl.

How to cite Zielska M, Lewandowski M, Widecka K, et al. uncommon com- plication of pulmonary arterial hypertension treatment with the parenteral use of  treprostinil. Kardiol Pol. 2021; 79: 79-80. doi:10.33963/KP.15672

referenceS

1 galiè N, Humbert M, Vachiery JL, et al. 2015 eSC/erS guidelines for the di- agnosis and treatment of pulmonary hypertension. eur Heart J. 2016; 37: 67-119.

2 Malaczynska -rajpold K, Kurzyna M, Koteja A, et al. The “bouncing” catheter. 

Cardiol J. 2016; 23: 552-553.

3 Desole S, Velik -Salchner C, Fraedrich g, et al. Subcutaneous implantation of  a new intravenous pump system for prostacyclin treatment in patients with pulmo- nary arterial hypertension. Heart Lung. 2012; 41: 599-605.

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