Key result
Recurrent compartment syndrome may rarely complicate heritable connective tissue disorders with EDS features.
Case Report (n=1)
Acute compartment syndrome may rarely present as a complication in patients with heritable connective tissue disorders beyond classical vascular Ehlers-Danlos syndrome.
Compartment syndrome warrants consideration in heritable connective tissue disorders beyond vEDS; case report leaves open need for prospective confirmation.
Arterial complications are common in vascular type Ehlers-Danlos syndrome (EDS), accounting for 66% of first complications. Several cases in the literature have documented acute compartment syndrome (ACS) following vascular rupture in vascular type EDS. Other disorders of connective tissue have also demonstrated vascular fragility, leading to arterial aneurysm and rupture, but there have been no documented cases of ACS. Here, we report on a female patient with a history of recurrent compartment syndrome who exhibits some clinical findings seen in hypermobile and vascular EDS; however she does not meet clinical and molecular diagnostic criteria for either of them. We further review the literature on ACS in heritable connective tissue disorders and suggest that compartment syndrome may rarely complicate other heritable disorders of connective tissue.
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Barajas et al. (2013) conducted a case report in Recurrent compartment syndrome and clinical features of a connective tissue disorder (n=1). A female patient with recurrent compartment syndrome exhibited clinical features of hypermobile and vascular EDS without meeting diagnostic criteria, suggesting compartment syndrome may rarely complicate other heritable connective tissue disorders.
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