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Vibrio Vulnificus Infection Presenting with Compartment Syndrome of Bilateral Calves
The Journal of Emergency Medicine, Vol. 35, No. 4, pp. 455– 457, 2008 Copyright © 2008 Elsevier Inc. Printed in the USA. All rights reserved 0736-4679/08 $–see front matter
Letters to the Editor e VIBRIO VULNIFICUS INFECTION PRESENTING WITH COMPARTMENT SYNDROME OF BILATERAL CALVES
pain completely. His left calf became increasingly firm, and redness developed on the left ankle; simultaneously, severe pain was elicited upon passive movement of his left leg. It was suspected that compartment syndrome was present caused by necrotizing fasciitis, and he underwent emergent fasciotomy and treatment with intravenous amoxicillin/clavulanic acid in the ED. Unfortunately, he ultimately required an above-knee amputation on the 4th day of hospitalization due to worsening of his condition despite treatment. In addition, an ecchymotic patch, swelling, and severe tenderness appeared on the right calf on the 3rd day of hospitalization (Figure 1). He required a fasciotomy on this extremity for necrotizing fasciitis and compartment syndrome, and three subsequent debridements of the right calf. Both the blood cultures and the wound cultures grew V. vulnificus, and antibiotics were adjusted to ceftriaxone disodium plus minocycline. He was discharged after 35 days of hospitalization, and it was not until later in his hospital course that he recalled he got an abrasion on his left calf from a piece of driftwood.
e To the Editor: Primary septicemia caused by Vibrio vulnificus infection is usually fulminant, rapidly progressive, and often leads to septic shock or multiple organ failure (1). It is seen in immunocompromised hosts and in patients with cirrhosis or hemochromatosis, but is rare with chronic renal failure. V. vulnificus septicemia manifesting with compartment syndrome of the lower extremities is rare. We report a young man who had V. vulnificus septicemia but presented with atypical features of fever and left calf pain initially thought to be a compartment syndrome, which, due to necrotizing fasciitis and bacteremia, spread to the right calf 3 days later. CASE REPORT A 29-year-old man came to our Emergency Department (ED) with fever, cough, sore throat, and left calf pain for 1 day. His medical history was significant for end-stage renal disease (ESRD) with regular hemodialysis, but he denied any history of diabetes mellitus, alcoholism, liver diseases, or trauma. Physical examination revealed an ill-appearing man with the following vital signs: temperature 37.6°C, respiratory rate 20 breaths/min, pulse 105 beats/min, and blood pressure 179/118 mm Hg. His left calf was warm, mildly swollen, and tender, but with no obvious wound. There was skin color change, bullae, and crepitation present over the involved leg, and the arterial pulse was decreased. Laboratory studies demonstrated a white blood cell count of 7200/mm3 (with a differential count of 93.5% segmented forms), platelet count 91,000/mm3, D-dimer 256 g/L (normal: ⬍ 250 g/L), glucose 126 mg/dL, aspartate transaminase 42 IU/L (normal: 10 –50 IU/L), creatinine 7.2 mg/dL, creatine phosphokinase 1401 IU/L (normal: 56 –244 IU/L), myoglobin ⬎ 10,000 g/L, and C-reactive protein 143.1 mg/L (normal: ⬍ 6 mg/L). Chest X-ray study and images of the left leg demonstrated normal findings and no subcutaneous gas. He was treated with narcotic analgesia but this failed to relieve his
DISCUSSION V. vulnificus is a Gram-negative, halophilic bacillus, and prefers low salinity and warm water temperature. The presentations of V. vulnificus infection may present as three syndromes: 1) primary bacteremia (sepsis with high fever but without an apparent focus of infection; history of consumption of raw oysters usually can be traced), 2) wound infection (direct injury from marine or preexisting skin wound with exposure to salt water, which may result in tissue necrosis, cellulitis, and necrotizing fasciitis), and 3) gastrointestinal illness (2). It is likely that our patient developed his symptoms after exposure to the driftwood, and the primary infection in his left calf resulted in bacteremia that seeded his right calf. Hemorrhagic bulla is the most common cutaneous manifestation of V. vulnificus infection, but variable presentations such as erythema, necrotizing fasciitis, vasculitis, purpura, gangrene, and urticaria have also been reported (3). Skin lesions usually appear on the extremities, and more than 90% occur on the legs. The time of the onset of symptoms after exposure is usually from hours to days (4). The occurrence of acute compartment syndrome associated with V. vulnificus infection is rare. In a large 455
456
Letters to the Editor
REFERENCES
Figure 1. The ecchymotic patches appeared over the right calf 3 days after admission.
retrospective study of V. vulnificus in Taiwan, 66.67% of the patients with cutaneous infection developed septic shock, and the mortality rate was high in patients with necrotizing fasciitis (40.5%) but lower in cases with cellulitis alone (6.7%). The overall mortality rate in this study was approximately 30% (5). Our case demonstrates some of the classic features associated with V. vulnificus infection with secondary bacteremia: the immunocompromised status (ESRD), fever, recent exposure to a marine environment from the driftwood, lower extremity pain, rapid progression to compartment syndrome (within 1 day), and fulminant bacteremia. Nonetheless, this case also can easily be mistaken for non-specific febrile illness with initial presentation of fever, cough, sore throat, and lower leg pain. CONCLUSIONS In conclusion, this case highlights that V. vulnificus septicemia with compartment syndrome should be suspected if the following features are present: fever, severe leg pain with or without purpuric skin lesions, recent exposure to a marine environment or consumption of raw seafood, and a past medical history of hepatic cirrhosis, hemochromatosis, or chronic renal failure.
Yu-Hui Lu, MD Department of Emergency Medicine Chi-Mei Medical Center, Liouying Tainan, Taiwan Hung-Jung Lin, MD, MBA Kuo-Chin Wu, MD Department of Emergency Medicine Chi-Mei Medical Center Tainan, Taiwan doi:10.1016/j.jemermed.2007.11.084
1. H’ng MW, Chew WY, Tan BK. Necrotizing fasciitis caused by Vibrio vulnificus: a review of four cases in a Singapore tertiary hospital. J Trauma 2005;59:482–5. 2. Hui KC, Zhang F, Komorowska-Timek E, et al. Compartment syndrome of the forearm as the initial symptom of systemic Vibrio vulnificus infection. J Hand Surg Am 1999;24:715–7. 3. Chiang SR, Chuang YC. Vibrio vulnificus infection: clinical manifestations, pathogenesis, and antimicrobial therapy. J Microbiol Immunol Infect 2003;36:81– 8. 4. Laughlin TJ, Lavery LA. Lower extremity manifestations of Vibrio vulnificus infection. J Foot Ankle Surg 1995;34:354 –7. 5. Park SD, Shon HS, Joh NJ. Vibrio vulnificus septicemia in Korea: clinical and epidemiologic findings in seventy patients. J Am Acad Dermatol 1991;24:397– 403.
e RETINOIC ACID SYNDROME SUBSEQUENT TO THE FIRST DOSE OF ALL TRANS RETINOIC ACID e To the Editor: We report a patient with retinoic acid syndrome (RAS), which presented after the first dose of all-trans retinoic acid (ATRA) in a patient with acute promyelocytic leukemia (APL), to increase awareness of this entity. ATRA was first used as a differentiating agent in 1987 in Shanghai for the treatment of APL (1). RAS became an important complication of the therapy after the administration of ATRA. RAS is defined as the presentation of any three of the following symptoms in the setting of ATRA administration: fever, weight gain, respiratory distress, interstitial pulmonary infiltrates, pleural and pericardial effusion, episodic hypotension, and acute renal failure. Early recognition and administration of dexamethasone are critical. There are few factors that predict the development of RAS. The white blood cell (WBC) count at presentation, the peak WBC count, or the rate of rise of the WBC may be predictive. The pathogenesis of the syndrome is poorly understood. Factors that might play a role are altered expressions of adhesion molecules on the surface of the differentiating APL blast and of the endothelial cells that may facilitate migration of the leukemic cells that cause damage to the tissue. The release of vasoactive substances and cytokines from the blast cells may contribute to the syndrome. Symptoms generally begin within the first 20 days of ATRA therapy (2).
CASE REPORT A 34-year-old woman presented to our clinic complaining of malaise, weakness, and gingival bleeding for a week. Body temperature was 37.5°C; vital signs were stable. On physical examination, mild conjunctival pal-
Letters to the Editor e VIBRIO VULNIFICUS INFECTION PRESENTING WITH COMPARTMENT SYNDROME OF BILATERAL CALVES
pain completely. His left calf became increasingly firm, and redness developed on the left ankle; simultaneously, severe pain was elicited upon passive movement of his left leg. It was suspected that compartment syndrome was present caused by necrotizing fasciitis, and he underwent emergent fasciotomy and treatment with intravenous amoxicillin/clavulanic acid in the ED. Unfortunately, he ultimately required an above-knee amputation on the 4th day of hospitalization due to worsening of his condition despite treatment. In addition, an ecchymotic patch, swelling, and severe tenderness appeared on the right calf on the 3rd day of hospitalization (Figure 1). He required a fasciotomy on this extremity for necrotizing fasciitis and compartment syndrome, and three subsequent debridements of the right calf. Both the blood cultures and the wound cultures grew V. vulnificus, and antibiotics were adjusted to ceftriaxone disodium plus minocycline. He was discharged after 35 days of hospitalization, and it was not until later in his hospital course that he recalled he got an abrasion on his left calf from a piece of driftwood.
e To the Editor: Primary septicemia caused by Vibrio vulnificus infection is usually fulminant, rapidly progressive, and often leads to septic shock or multiple organ failure (1). It is seen in immunocompromised hosts and in patients with cirrhosis or hemochromatosis, but is rare with chronic renal failure. V. vulnificus septicemia manifesting with compartment syndrome of the lower extremities is rare. We report a young man who had V. vulnificus septicemia but presented with atypical features of fever and left calf pain initially thought to be a compartment syndrome, which, due to necrotizing fasciitis and bacteremia, spread to the right calf 3 days later. CASE REPORT A 29-year-old man came to our Emergency Department (ED) with fever, cough, sore throat, and left calf pain for 1 day. His medical history was significant for end-stage renal disease (ESRD) with regular hemodialysis, but he denied any history of diabetes mellitus, alcoholism, liver diseases, or trauma. Physical examination revealed an ill-appearing man with the following vital signs: temperature 37.6°C, respiratory rate 20 breaths/min, pulse 105 beats/min, and blood pressure 179/118 mm Hg. His left calf was warm, mildly swollen, and tender, but with no obvious wound. There was skin color change, bullae, and crepitation present over the involved leg, and the arterial pulse was decreased. Laboratory studies demonstrated a white blood cell count of 7200/mm3 (with a differential count of 93.5% segmented forms), platelet count 91,000/mm3, D-dimer 256 g/L (normal: ⬍ 250 g/L), glucose 126 mg/dL, aspartate transaminase 42 IU/L (normal: 10 –50 IU/L), creatinine 7.2 mg/dL, creatine phosphokinase 1401 IU/L (normal: 56 –244 IU/L), myoglobin ⬎ 10,000 g/L, and C-reactive protein 143.1 mg/L (normal: ⬍ 6 mg/L). Chest X-ray study and images of the left leg demonstrated normal findings and no subcutaneous gas. He was treated with narcotic analgesia but this failed to relieve his
DISCUSSION V. vulnificus is a Gram-negative, halophilic bacillus, and prefers low salinity and warm water temperature. The presentations of V. vulnificus infection may present as three syndromes: 1) primary bacteremia (sepsis with high fever but without an apparent focus of infection; history of consumption of raw oysters usually can be traced), 2) wound infection (direct injury from marine or preexisting skin wound with exposure to salt water, which may result in tissue necrosis, cellulitis, and necrotizing fasciitis), and 3) gastrointestinal illness (2). It is likely that our patient developed his symptoms after exposure to the driftwood, and the primary infection in his left calf resulted in bacteremia that seeded his right calf. Hemorrhagic bulla is the most common cutaneous manifestation of V. vulnificus infection, but variable presentations such as erythema, necrotizing fasciitis, vasculitis, purpura, gangrene, and urticaria have also been reported (3). Skin lesions usually appear on the extremities, and more than 90% occur on the legs. The time of the onset of symptoms after exposure is usually from hours to days (4). The occurrence of acute compartment syndrome associated with V. vulnificus infection is rare. In a large 455
456
Letters to the Editor
REFERENCES
Figure 1. The ecchymotic patches appeared over the right calf 3 days after admission.
retrospective study of V. vulnificus in Taiwan, 66.67% of the patients with cutaneous infection developed septic shock, and the mortality rate was high in patients with necrotizing fasciitis (40.5%) but lower in cases with cellulitis alone (6.7%). The overall mortality rate in this study was approximately 30% (5). Our case demonstrates some of the classic features associated with V. vulnificus infection with secondary bacteremia: the immunocompromised status (ESRD), fever, recent exposure to a marine environment from the driftwood, lower extremity pain, rapid progression to compartment syndrome (within 1 day), and fulminant bacteremia. Nonetheless, this case also can easily be mistaken for non-specific febrile illness with initial presentation of fever, cough, sore throat, and lower leg pain. CONCLUSIONS In conclusion, this case highlights that V. vulnificus septicemia with compartment syndrome should be suspected if the following features are present: fever, severe leg pain with or without purpuric skin lesions, recent exposure to a marine environment or consumption of raw seafood, and a past medical history of hepatic cirrhosis, hemochromatosis, or chronic renal failure.
Yu-Hui Lu, MD Department of Emergency Medicine Chi-Mei Medical Center, Liouying Tainan, Taiwan Hung-Jung Lin, MD, MBA Kuo-Chin Wu, MD Department of Emergency Medicine Chi-Mei Medical Center Tainan, Taiwan doi:10.1016/j.jemermed.2007.11.084
1. H’ng MW, Chew WY, Tan BK. Necrotizing fasciitis caused by Vibrio vulnificus: a review of four cases in a Singapore tertiary hospital. J Trauma 2005;59:482–5. 2. Hui KC, Zhang F, Komorowska-Timek E, et al. Compartment syndrome of the forearm as the initial symptom of systemic Vibrio vulnificus infection. J Hand Surg Am 1999;24:715–7. 3. Chiang SR, Chuang YC. Vibrio vulnificus infection: clinical manifestations, pathogenesis, and antimicrobial therapy. J Microbiol Immunol Infect 2003;36:81– 8. 4. Laughlin TJ, Lavery LA. Lower extremity manifestations of Vibrio vulnificus infection. J Foot Ankle Surg 1995;34:354 –7. 5. Park SD, Shon HS, Joh NJ. Vibrio vulnificus septicemia in Korea: clinical and epidemiologic findings in seventy patients. J Am Acad Dermatol 1991;24:397– 403.
e RETINOIC ACID SYNDROME SUBSEQUENT TO THE FIRST DOSE OF ALL TRANS RETINOIC ACID e To the Editor: We report a patient with retinoic acid syndrome (RAS), which presented after the first dose of all-trans retinoic acid (ATRA) in a patient with acute promyelocytic leukemia (APL), to increase awareness of this entity. ATRA was first used as a differentiating agent in 1987 in Shanghai for the treatment of APL (1). RAS became an important complication of the therapy after the administration of ATRA. RAS is defined as the presentation of any three of the following symptoms in the setting of ATRA administration: fever, weight gain, respiratory distress, interstitial pulmonary infiltrates, pleural and pericardial effusion, episodic hypotension, and acute renal failure. Early recognition and administration of dexamethasone are critical. There are few factors that predict the development of RAS. The white blood cell (WBC) count at presentation, the peak WBC count, or the rate of rise of the WBC may be predictive. The pathogenesis of the syndrome is poorly understood. Factors that might play a role are altered expressions of adhesion molecules on the surface of the differentiating APL blast and of the endothelial cells that may facilitate migration of the leukemic cells that cause damage to the tissue. The release of vasoactive substances and cytokines from the blast cells may contribute to the syndrome. Symptoms generally begin within the first 20 days of ATRA therapy (2).
CASE REPORT A 34-year-old woman presented to our clinic complaining of malaise, weakness, and gingival bleeding for a week. Body temperature was 37.5°C; vital signs were stable. On physical examination, mild conjunctival pal-