Formosan Journal of Surgery (2014) 47, 62e65

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CASE REPORT

Amputation of lower limb for necrotizingsoft-tissue infection in an ultramarathonrunner

Yu-Hao Huang a,b, Tung-Ying Hsieh a,b, I-Chien Chen b,Chung-Sheng Lai b,c, Sin-Daw Lin b,c, Su-Shin Lee b,c,Kao-Ping Chang b,c,*

aDepartment of Surgery, Kaohsiung Medical University Hospital, Kaohsiung, TaiwanbDivision of Plastic and Reconstructive Surgery, Department of Surgery,Kaohsiung Medical University Hospital, Kaohsiung, Taiwanc Faculty of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan

Received 23 July 2013; received in revised form 29 August 2013; accepted 4 October 2013Available online 3 January 2014

KEYWORDSathlete;marathon;necrotizing infection

Conflicts of interest: All authors d* Corresponding author. Division of P

Tzyou 1st Road, Kaohsiung 807, TaiwaE-mail address: kapich@kmu.edu.t

1682-606X/$ - see front matter Copyrhttp://dx.doi.org/10.1016/j.fjs.2013.

Summary Necrotizing soft-tissue infection (NSTI) is a life-threatening disease with rapid pro-gression, which has rarely been discussed in the medical literature with regard to marathonrunners. We present the case of a 51-year-old Taiwanese woman, a female ultramarathon run-ner who had a medical disaster after her participation in La Trans-Gaule French ultramarathon.After completing the competition, she was diagnosed to have septicemia and a potentially life-threatening NSTI of both lower limbs. Therefore, she underwent emergent right above-the-knee amputation and left foot transmetatarsal amputation. Then she came back to Taiwanto receive further treatment. After meticulous reconstructive surgery and continuous rehabil-itation programs, she was instructed to wear prostheses and finally resumed her daily activ-ities. Although several organ systems can be affected by marathon running, soft-tissueinfections have seldom been discussed. In our case, the patient suffered from bilateral lowerlimb amputation caused by severe necrotizing infection after the competition. Thus, soft-tissue infection is also an important issue for ultramarathon runners and medical service pro-viders from ultramarathon associations.Copyright ª 2013, Taiwan Surgical Association. Published by Elsevier Taiwan LLC. All rightsreserved.

eclare no conflicts of interest.lastic and Reconstructive Surgery, Department of Surgery, Kaohsiung Medical University Hospital, 100n.w (K.-P. Chang).

ight ª 2013, Taiwan Surgical Association. Published by Elsevier Taiwan LLC. All rights reserved.10.003

Amputation for necrotizing soft-tissue infection 63

1. Introduction

ischemic bilateral foot. Some toes were erythematous and

Marathon running has been a popular sport around theworld. Medical problems of the musculoskeletal, gastroin-testinal, cardiac, renal, and pulmonary systems and elec-trolyte and fluid imbalance in marathon runners havewidely been discussed.1 Environmental factors also play animportant role in the patients’ needs of medical attentionin a marathon race.1 After searching PubMed, we havefound that there is still no article discussing the relationshipbetween marathon and soft-tissue infection to date.Necrotizing soft-tissue infections (NSTIs) have highmorbidity and mortality rates and should be diagnosed andmanaged as early as possible.2,3 Here, we present an un-usual case of a patient who suffered from a devastatingseptic shock and underwent lower limb amputationsbecause of NSTIs after a marathon race.

2. Case report

This 51-year-old woman took part in an ultramarathon race,La Trans-Gaule. She was a talented Taiwanese marathonrunner and had been nicknamed “ultramarathon mama.”She had to run almost 50e60 km every day in the compe-tition. After completing the race, she visited the emer-gency department of a local hospital due to severe pain inboth lower legs. At the emergency department, she wasafebrile, but blisters on both feet associated with mildswelling were noted. The vital signs on arrival were rela-tively stable. The hemogram showed leukopenia and ane-mia. Serum biochemical analysis showed elevation of C-reactive protein, hyponatremia, and impairment of liverfunction, as shown in Table 1. She was then admitted underthe impression of lower limb cellulitis and mild dehydra-tion. After admission, she was treated with broad-spectrumantibiotics and intravenous fluid.

Unexpectedly, she was found in shock status the nextmorning. There were ecchymoses and mottling of the skinextending from both feet to middle calves. Bilateral tensionof the feet skin texture with poor capillary refilling wasnoted. Moreover, follow-up blood tests revealed progres-sively worsening data. With the deteriorating status of thepatient, she was immediately transferred to a medicalcenter hospital.

At the medical center hospital, she appeared ill andlethargic. Her blood pressure dropped to 90/60 mmHg.

Table 1 Initial laboratory work.

Result Normal

White blood cells (mm3) 1120 4000e10,500Hemoglobin (g/dL) 10.4 11.9e15.5 for femalePlatelets (mm3) 197,000 150,000e450,000C-reactive protein (mg/L) 400 <5.0ALT/AST (IU/L) 178/85 10e42/10e40BUN/Cr (mg/dL) 2/0.9 7e18/0.6e1.3Na (mmol/L) 129 136e145

ALT Z alanine aminotransferase; AST Z aspartate amino-transferase; BUN Z blood urea nitrogen.

Bilateral skin necroses were noted on the cold, partial

swollen with pus formation. There were several large blis-ters on both cyanotic heels. Poor pulsation of the dorsalispedis artery was noted bilaterally. Repeated laboratoryresults are shown in Table 2. The hemogram showed severeleukopenia and high levels of creatine kinase and lactate.The Laboratory Risk Indicator for Necrotizing Fasciitis(LRINEC) score was calculated to be 8, which strongly sug-gested necrotizing fasciitis (NF). Finally, she was diagnosedto have bilateral NSTI of lower legs combined withcompartment syndrome and septic shock.

The runner was hospitalized in a medical center hospitalin France for 27 days. She underwent emergent right above-the-knee amputation and left foot transmetatarsal ampu-tation. As for the infection and shock status, antibiotics andinotropes were administered. In the following days, hercondition was gradually stabilized. Besides, bacterial cul-tures yielded group A streptococcus and methicillin-sensitive Staphylococcus aureus. The other woundsbecame stable with the use of broad-spectrum antibioticsand vacuum-based dressing. Finally, the patient wastransferred back to a medical center in Taiwan for follow-up care.

On arrival, the vital signs were relatively stable exceptfor mild fever. The right amputation stump was wellhealed and the left leg was covered with full-thicknessskin defect with odorous and devitalized tissues. TheAchilles tendon and the flexor hallucis longus tendonwere exposed as shown in Fig. 1. After obtaining the re-sults of wound culture, adequate fluid resuscitation andempirical antibiotics were started. After admission,regional fasciotomy and debridement of the left leg wereperformed on the next day. The wounds were treatedwith frequent dressing changes, broad-spectrum antibi-otics, and hyperbaric oxygen therapy. Afterward,debridement and skin grafting were consecutively per-formed. She practiced wheelchair ambulation and thenshifted to a walker with a right transfemoral prosthesisand left prosthetic foot smoothly. As her general andwound conditions improved, she was discharged 2 monthslater with regular outpatient clinic follow-up (Fig. 2). Thepatient is now wearing bilateral prostheses and has freeambulation (Fig. 3).

Table 2 Laboratory examinations the next day.

Result Normal

White blood cells (mm3) 640 4000e10,500Hemoglobin (g/dL) 9.8 11.9e15.5 for femalePlatelets (mm3) 79,000 150,000e450,000C-reactive protein (mg/L) 329 <5.0BUN/Cr (mg/dL) 31/0.8 7e18/0.6e1.3Na (mmol/L) 126 136e145CK (U/L)/fibrinogen (g/L) 1976/7 25e145/2e4pH/bicarbonates (mmol/L) 7.36/16 7.35e7.45/22e26Lactate (mg/dL) 54 <5Glucose (mg/dL) 158 70e130

BUN Z blood urea nitrogen; CK Z creatine kinase.

Figure 1 Initial presentation in Taiwan, nearly 1 month afterthe injury. Results of the right lower extremity above-the-kneeamputation and left lower extremity transmetatarsal ampu-tation, with skin defect over 16% of total body surface area.

Figure 3 Eleven months after the injury. This ultramarathonrunner can have free ambulation with suitable prostheses, andshe states that she hopes to resume running in the future.

64 Y.-H. Huang et al.

3. Discussion

Several organ systems can be affected by marathonrunning.1 Most problems that have been mentioned involvethe musculoskeletal, gastrointestinal, cardiac, renal, andpulmonary systems, and also include electrolyte and fluidimbalance, but seldom soft-tissue infections. Becausemarathon has become a popular sport, many medical issuesare discussed to prevent morbidity and mortality.1,4,5 Ofmarathon participants, 2e8% will seek medical attentionduring or immediately after completing the race.5,6 Someof the problems are self-limited and easily solved, but someare life threatening, such as severe dehydration, hypona-tremia, heat stroke, acute renal failure, acute coronarysyndrome, and ischemic colitis.1 Environmental factors arealso proved to play an important role in causing the run-ners’ needs for medical service.1

Figure 2 Four months after the injury. The wound has beendebrided once, and skin grafting was performed twice. Thewound healing is satisfactory.

However, soft-tissue infections in marathon races wereseldom discussed in the past, and in fact no medical articleswere found after our PubMed search using the keywords“marathon” and “soft tissue infection.” In our opinion,most marathon runners have ignored the skin wounds,regarding them as nothing special. Marathon runners,however, should protect the developing bullae from beingbroken and care the wound, if necessary, with occlusivedressing. Ultramarathon, an even more strenuous compe-tition combined with a critical environment, may causemultiple skin breaks more frequently, and thus increase thepossibility of soft-tissue infections.7 Combined with adehydration status and relative ischemia of muscles causedby extreme straining, the condition might progress quicklyto disseminated NSTI. In this unusual case, the patientnoticed only common, small blisters on her feet initially.However, she collapsed on the last day after completing thecontest because of severe sepsis caused by the NSTI andcompartment syndrome of her legs. She subsequently un-derwent major amputation to control the infection. If theNSTI had been detected earlier by the runner or first-linemedical service providers from the ultramarathon associa-tion, this kind of tragedy would have been avoided in thisextremely healthy marathon runner. A lack of medical aidsin the long critical race may cause further deteriorations.Thus, the importance of soft-tissue care should also beemphasized to the ultramarathon runners and ultramara-thon association.

Table 3 Laboratory risk indicator for necrotizing fasciitis(LRINEC) score.

b Score

C-reactive protein, mg/L<150 0 0�150 3.5 4

Total white cell count, per mm3

<15 0 015e25 0.5 1>25 2.1 2

Hemoglobin, g/dL>13.5 0 011e13.5 0.6 1<11 1.8 2

Sodium, mmol/L�135 0 0<135 1.8 2

Creatinine, mmol/L�141 0 0>141 1.8 2

Glucose, mmol/L�10 0 0>10 1.2 1

LRINECZ Z Laboratory Risk Indicator for Necrotizing Fasciitis,�6 on the LRINEC score strongly suggests necrotizing fasciitis.

Amputation for necrotizing soft-tissue infection 65

NSTI is notorious for its rapid progression, extensivedestruction of tissue, systemic toxicity, and even loss oflimbs.5,8,9 NSTIs are clinically manifested by fulminantdestruction of tissue, signs of systematic toxicity, and ahigh rate of mortality.3,10,11 There are an estimated 3.5cases/100,000 persons with a case-fatality rate of 24% forNSTIs.12 Clinical manifestations include systemic findings,such as fever, tachycardia, and hypotension, and typicallocal signs and symptoms such as tense edema outside theinvolved skin, disproportionate pain, blisters/bullae, crep-itus, and subcutaneous gas.3 Early recognition of NSTIs isimportant. Wong et al13 proposed a scoring system, termedthe LRINEC score, which uses a combination of abnormallaboratory variables to calculate the risk of NSTIs (Table 3).If the score is 6 or higher, the conditions are highly pre-dictive of NF, with a positive predictive rate of about 98%and a negative predictive value of about 96%. The score ofour patient upon arrival to the local hospital was 8, whichwas indicative of NSTI.

If NSTI is recognized, urgent and aggressive managementis mandatory. The keys are to explore and remove thenecrotic infected tissue, to restore tissue perfusion by he-modynamic support, and to administer broad-spectrumantimicrobial therapy. A delay in surgical diagnosis anddebridement increases the rates of severe morbidity andmortality.10 To salvage the septic shock status in our case,immediate amputation of the infected limbs was decided.The tragedy suffered by our patient will be a valuablelesson to all the marathon runners, and the ultramarathonassociation should pay due medical attention to soft-tissueinfections. Therefore, it is necessary to remind the runnersof maintaining adequate hydration and instruct them not toignore their skin lesions.

Marathon running has been a popular sport worldwide.Several organ systems have been affected by marathonrunning, including the musculoskeletal, gastrointestinal,cardiac, renal, and pulmonary systems, and the runnersalso experience electrolyte and fluid imbalance. AlthoughNSTIs are rarely encountered in marathon races, a failure inearly diagnosis may lead to significant morbidity and evenmortality. In our opinion, NSTIs should also be considered asan important issue in ultramarathon races, and medicalservice providers from the ultramarathon associationshould pay due attention to this problem.

Acknowledgments

Special thanks go to the doctors in France for their imme-diate and appropriate first aid given to the marathonrunner.

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