Multifocal recurrent atraumatic acute compartment syndrome in an adolescent

Matthew A. Majer; Jaxon Boley; Jennifer Acevedo; Fengju Xun; Angel F. San Roman

doi:10.18203/issn.2455-4510.IntJResOrthop20263947

Authors

Matthew A. Majer American University of the Caribbean, Cupecoy, Sint Maarten
Jaxon Boley American University of the Caribbean, Cupecoy, Sint Maarten
Jennifer Acevedo St. Georges University, University Centre Grenada, West Indies, Grenada
Fengju Xun American University of the Caribbean, Cupecoy, Sint Maarten
Angel F. San Roman Angel F. San Roman Paediatrics, Coral Gables, FL

DOI:

https://doi.org/10.18203/issn.2455-4510.IntJResOrthop20263947

Keywords:

Compartment syndrome, Atraumatic, Recurrent, Fasciotomy, Vascular malformation, Adolescent

Abstract

Recurrent, atraumatic acute compartment syndrome (ACS) is an uncommon and poorly understood phenomenon. We report a female in her teens who experienced two distinct episodes of atraumatic ACS. The first episode involved the right lower extremity, and the second, the right upper extremity. Each case required emergent fasciotomy and involved postoperative complications, requiring repeat surgical intervention and vacuum-assisted closure. Extensive multidisciplinary evaluation, including rheumatologic, hematologic, and genetic testing, failed to identify an etiology. Given the recurrence and localization to separate limbs, a focal vascular abnormality is suspected, supported by comparison with a similar reported case successfully treated with angiographic embolization. This case underscores the importance of prompt surgical intervention and multidisciplinary evaluation and highlights the potential role of vascular malformations in cases of unexplained compartment syndrome.

References

Tuckey J. Bilateral compartment syndrome complicating prolonged lithotomy position. Br J Anaesth. 1996;77:546-9.

Blaisdell FW. The pathophysiology of skeletal muscle ischemia and the reperfusion syndrome: a review. Cardiovasc Surg. 2002;10:620-30.

Raza H, Mahapatra A. Acute compartment syndrome in orthopedics: causes, diagnosis, and management. Adv Orthop. 2015;2015:543412.

Torlincasi AM, Lopez RA, Waseem M. Acute compartment syndrome. National Center for Biotechnology Information. Treasure Island, FL: StatPearls Publishing. 2025.

Donaldson J, Goddard N. Compartment syndrome in patients with haemophilia. J Orthop. 2015;12:237-41.

Calf strain. Physiopedia. 2025. Available at: https://www.physiopedia.com/index.php?title=Calf_Strain&oldid=367184. Accessed on 13 February 2026.

Roache-Robinson P, Killeen RB, Hotwagner DT. IgA vasculitis (Henoch-Schönlein purpura). In: StatPearls. Treasure Island, FL: StatPearls Publishing. 2025.

McQueen MM, Gaston P, Court-Brown CM. Acute compartment syndrome. Who is at risk? J Bone Joint Surg Br. 2000;82(2):200-3.

Duckworth AD, McQueen MM. The diagnosis of acute compartment syndrome: a critical analysis review. JBJS Rev. 2017;5(12):e1.

McQueen MM, Court-Brown CM. Compartment monitoring in tibial fractures. The pressure threshold for decompression. J Bone Joint Surg Br. 1996;78(1):99-104.

Ogrodnik J, Oliver JD, Cani D, Boczar D, Huayllani MT, Restrepo DJ, et al. Clinical case of acute non-traumatic hand compartment syndrome and systematic review for the upper extremity. Hand (N Y). 2021;16(3):285-91.

Ebeling M, Hudson B. Recurrent, atraumatic acute compartment syndrome secondary to IgA vasculitis: a case report. J Am Coll Emerg Physicians Open. 2024;5(4):e13270.

Shiraz A, Bakhtiar H, Farman G, Khan S, Rafiq N. Unidentified recurrent acute compartment syndrome of the right upper limb. Cureus. 2022;14(2):e22033.

Dalton DM, Munigangaiah S, Subramaniam T, McCabe JP. Acute bilateral spontaneous forearm compartment syndrome. Hand Surg. 2014;19(1):99-102.

Arbeloa-Gutierrez L, Arenas-Miquelez A, Muñoa L, Gordillo A, Eslava E, Insausti I, et al. Lateral circumflex femoral artery false aneurysm as a complication of intertrochanteric hip fracture with displaced lesser trochanter. J Surg Case Rep. 2019;2019(6):rjz184.

Livingston KS, Meehan WP 3rd, Hresko MT, Matheney TH, Shore BJ. Acute exertional compartment syndrome in young athletes: a descriptive case series and review of the literature. Pediatr Emerg Care. 2018;34(2):76-80.