Drug-induced angioedema leads to rare condition in woman: Report

Medication cocktail caused compartment syndrome affecting all limbs in case

Marisa Wexler, MS avatar

by Marisa Wexler, MS |

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A large number of oral medications are seen scattered together alongside two prescription pill bottles.

The unusual case of a woman with drug-induced angioedema that led to a rare complication called acute compartment syndrome (ACS) in all four of her limbs was described in a recent report from Japan.

ACS is a disorder in which the pressure within one of the body’s compartments — one of the sections of the body, such as a limb or the abdominal cavity — becomes so high that blood can’t flow through blood vessels. This can lead to tissue death, as cells in the affected area are starved of oxygen and nutrients.

The condition is not often seen with the rare swelling disorder.

Indeed, according to the researchers, “the emergence of acute extremity [limb] compartment syndrome secondary to angio-oedema is exceedingly rare.”

Their report, “Acute quadruple extremity compartment syndrome due to angio-oedema after polypharmacy overdose including olmesartan medoxomil, telmisartan and vildagliptin,”  was published in BMJ Case Reports.

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Angioedema is characterized by swelling taking place in the deeper layers of the skin or in the mucus membranes. Some medications can trigger angioedema as a side effect, causing what is known as drug-induced or nonallergic angioedema.

There are several known complications that can occur due to angioedema, including airway obstruction, which can happen when swelling occurs in the tongue or throat. In rare cases, acute compartment syndrome can develop as a complication.

In this study, a pair of researchers from the Kameda Medical Center, in Japan, reported the case of a woman in her 50s who was brought to the emergency department after overdosing on a cocktail of different medications. These included two different angiotensin receptor blockers (ARBs), which are a class of medicines frequently used to treat high blood pressure; these drugs have been associated with the development of nonallergic angioedema.

Upon her admission to the hospital, the woman was unconscious with very low blood pressure. She was taken to an intensive care unit for supportive treatment.

About a day later, doctors noticed swelling in her limbs, without any other apparent changes. A day after that, the woman regained consciousness and reported pain in her hand.

Her doctors’ examinations showed “significant tension in all limbs,” with her hands showing signs of blistering and necrosis, or tissue death.

Additional diagnostic tests showed increased pressure in all four of her limbs, confirming the diagnosis of ACS. Based on the timing of the acute compartment syndrome in relation to her overdose and the specific medications involved, including ARBs, her official diagnosis was judged to be ACS resulting from drug-induced angioedema.

“Extensive diagnostic work and comprehensive clinical evaluation firmly established the cause of the [angioedema] as pharmacologically induced,” the researchers wrote.

The patient’s ACS was treated with a surgical procedure called fasciotomy, which basically involves making a large incision in the affected compartment to relieve pressure.

“Prompt recognition and intervention in cases of ACS, especially via fasciotomy, are critical,” the researchers noted.

This scenario is a very rare case caused by drug-induced [angioedema], which underscores the importance of vigilant monitoring to detect ACS in patients with progressing limb [swelling].

She was discharged from the hospital 58 days after being admitted, and engaged in physical therapy for six weeks after that.

According to the team, she has generally recovered well and is able to perform day-to-day activities independently, though she was still reporting some numbness and limited mobility in her hands two months after the fasciotomy.

The researchers noted that drug-induced angioedema “typically presents without the classic signs of allergic reactions, such as hives.” As such, it is key that patients are followed closely to ensure prompt diagnosis and treatment.

“This scenario is a very rare case caused by drug-induced [angioedema], which underscores the importance of vigilant monitoring to detect ACS in patients with progressing limb [swelling],” the team wrote.