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• W2008015488 abstract "Streptococcus pyogenes [group A Streptococcus (GAS)] causes a wide spectrum of invasive disease including toxic shock syndrome, bacteremia and tissue-destroying processes such as necrotizing fasciitis and myonecrosis.1-3 We report a case of severe invasive GAS disease in a child whose leukemic condition may have favorably influenced the initial clinical course. Case report. A previously healthy 3-year-old white boy presented to his primary physician with malaise, fever, coryza, sore throat and pain in the right knee. The child was ill-appearing and pale and had erythematous enlarged tonsils. A rapid streptococcal identification test of a pharyngeal swab sample was positive. The child was sent to our regional emergency department for further diagnostic tests and treatment. While en route to the hospital the patient's clinical condition deteriorated rapidly. On arrival at the emergency department, he had a temperature of 38.7°C, a respiratory rate of 40 breaths/min, a heart rate of 166 beats/min and a systolic blood pressure of 72 mm Hg. The patient alternated between obtundation and confusion. His mucous membranes were pale. The skin exhibited a diffuse and rapidly progressive petechial rash and a prolonged capillary refill time. Examination of the right knee was unremarkable. Epistaxis and gingival bleeding were evident, as was hepatomegaly. Blood cultures were obtained before the patient received intravenous doses of vancomycin and ceftazidime. Because of shock and impending cardiorespiratory collapse the child was intubated and received aggressive fluid resuscitation before transfer to the intensive care unit. Laboratory measurements on admission included a white blood cell count of 1.0 × 109/l with 23% band forms, 15% segmented forms, 50% lymphocytes, 11% monocytes, 1% eosinophils and no blasts seen on peripheral smear; hemoglobin of 8.0 g/dl; platelet count of 10 × 109/l; serum sodium of 127 mEq/l; and serum creatinine of 79.2 μmol/l (0.9 mg/dl). Arterial blood gas analysis during positive pressure ventilation revealed compensated metabolic acidosis. During the first 24 h of admission the patient required fluid resuscitation and infusions of epinephrine, dopamine and dobutamine to maintain adequate cardiovascular status. The child developed acute respiratory distress syndrome requiring significant ventilatory support. A bone marrow sample obtained by aspiration revealed features of pre-B cell acute lymphocytic leukemia. On Day 2 of hospitalization the patient developed swelling of the right calf. Computerized axial tomogram of the right lower extremity revealed an enlarged soleus muscle with homogeneous low attenuation and no indication of abscess formation. The diagnosis of myositis was supported by a serum creatinine phosphokinase (CPK) level of 2297 IU/l (normal, 20 to 240 IU/l). Intravenous penicillin was added to the antibiotic regimen after Gram-positive cocci in pairs and chains were identified in the initial blood culture. Further identification revealed the organism to be a beta-hemolytic group A Streptococcus (GAS). During Days 3 and 4 of hospitalization, the child continued to have fever and increasing edema of the right calf. In addition there was a purple discoloration of the right calf and thigh surrounded by erythema. Compartment pressures of the distal lower extremity were within acceptable range. One milliliter of serous fluid was obtained for bacteriologic examination by needle puncture of the lateral fluid collection and showed no organism. Vancomycin treatment was discontinued and intravenous clindamycin treatment was started. Intravenous immunoglobulin (1 g/kg) was also administered. Magnetic resonance imaging of the right lower extremity obtained on Day 5 of hospitalization showed severe circumferential edema of the subcutaneous fatty tissues and severe edema of the soleus muscle with marked contrast enhancement. Surgical exploration confirmed marked subcutaneous edema but no evidence of muscle necrosis or abscess formation. Biopsy specimens were obtained (Fig. 1, a and b) and revealed a mild inflammatory myopathy. Gram stain and cultures of biopsy specimens failed to identify any organism. The patient was started on chemotherapy for leukemia. The patient maintained a white blood cell count below 5.0 × 109/l during hospital admission as a result of the chemotherapy treatment.Fig. 1: a and b, first muscle biopsy revealing inflammatory myopathy with mild mononuclear infiltrate; c and d, second muscle biopsy revealing acute suppurative myositis with coagulative muscle necrosis. Hematoxylin and eosin. a and b, ×40; c and d, ×200.The patient completed a 10-day course of penicillin and clindamycin. Ceftazidime treatment and trimethoprim-sulfamethoxazole prophylaxis were continued because of neutropenia. He was extubated on Day 12 of hospitalization. By hospital discharge on Day 28 he had complete resolution of the process involving the right lower extremity. Sixteen days after hospital discharge the patient had a white blood cell count of 15.3 × 109/l, and 2 days later he developed increasing tenderness over the right calf area and inability to ambulate. Diffuse edema and erythema were present over the right calf with an indurated mass involving the gastrocnemius and soleus muscle complex measuring 8 by 10 cm. Surgical exploration and biopsy of the involved muscles were done. Biopsy specimens were obtained form the same muscle groups involved in the first episode of myositis. Histologic examination of the biopsy specimens revealed extensive myonecrosis with dense neutrophilic infiltrate and edema (Fig. 1, c and d). Cefazolin treatment was administered for 48 h until culture specimens (blood, throat and surgical wound) were negative. He had significant clinical improvement during the ensuing 4 days and was discharged home. On follow-up examination 1 week later the patient had normal healing of the surgical wound and complete resolution of clinical signs and symptoms. He has now completed the chemotherapy regimen, and the leukemia is in remission at the 1-year follow-up visit. Discussion. Group A streptococci are a rare cause of myositis, involving mostly adult patients in tropical regions.4 Only 21 cases of streptococcal myositis or myonecrosis were reported between 1900 and 19855; and 5 additional cases have been described in more recent reports.6-8 Streptococcal myositis is associated with a wide clinical spectrum, ranging from a subacute localized form with good prognosis to a serious acute presentation involving rapidly progressive disease, shock, multiple organ failure and a high case fatality rate (∼80 to 100%).9 Our patient had the more serious form of the disease, having presented with bacteremia, shock and respiratory failure. Although intense pain may be the only presenting symptom, the diagnosis of streptococcal myositis is generally suggested by the presence of swelling of the involved muscle tissue, fever and cutaneous alterations such as erythema or purple discoloration, petechiae or vesicles.10 Ultrasonography, computerized axial tomography and magnetic resonance imaging can be valuable adjuncts to confirming the diagnosis by revealing edema of muscle tissue, abscess formation and inflammation.4 A raised serum CPK can also be helpful in supporting the diagnosis.8 Necrotizing fasciitis, a more common form of invasive GAS disease, can generally be distinguished from streptococcal myositis during surgical exploration. However, these two clinical conditions have overlapping characteristics and may be present simultaneously in the same patient.5 There does not seem to be a difference in virulence between the GAS causing acute pharyngitis and those that cause severe invasive disease.11 In fact we speculate that our patient acquired GAS by the pharyngeal route as suggested by the early presenting symptoms and a positive rapid streptococcal detection test. After invasion the organism produces exotoxin A and/or B,2, 12 which ultimately leads to tissue destruction. GAS exotoxins are associated with the severity of invasive disease,12 presumably by functioning as superantigens capable of stimulating the synthesis of proinflammatory cytokines like tumor necrosis factor alpha, interleukin 1 beta and interleukin 6 from a variety of cell types.13-15 Our patient presented initially with invasive GAS infection in conjunction with severe leukopenia as result of new onset leukemia. The diagnosis of streptococcal myonecrosis was inferred by the presence of localized tenderness and swelling of the right calf, fever, elevated serum CPK and edema of the soleus muscle demonstrated by imaging studies. It was intriguing, however, that the initial surgical exploration revealed only mild edema with absence of necrotic tissue. Furthermore histology of biopsy specimens revealed fairly normal muscle tissue with a mild degree of edema and minimal cellular infiltration. When our patient presented nearly 3 weeks later with pain, functional loss, edema and cutaneous discoloration over the right calf, a repeat surgical exploration was promptly performed. The dense cellular infiltrate within the soleus muscle and surrounding tissues despite the absence of a bacterial pathogen was once again intriguing. The absence of GAS at the site of necrosis suggests that the mechanism of tissue destruction was host-mediated and not resurgence of a bacterial infection. We speculate that the original episode of soleus myositis caused by GAS promoted changes in the muscle, presumably by production of proinflammatory cytokines and chemoattractants.13-15 The patient's inability to produce competent circulating leukocytes at presentation resulted in only limited cellular infiltration and tissue damage to the soleus muscle and surrounding tissues. Days later, when the chemotherapy-mediated bone marrow suppression had resolved and circulating cells had regained normal function,16 there was significant leukocyte mobilization at the site of original injury with evidence of sterile inflammation and tissue injury involving the right calf. This phenomenon has been well-described in the pulmonary literature where acute lung injury is markedly attenuated in animals subjected to granulocyte depletion by chemotherapy agents before a noxious insult.17 However, a recent report by Kuo et al.18 suggests that mice infected with protease-positive (exotoxin B) GAS strains can suffer direct tissue injury, even when circulating neutrophils are depleted by vinblastine treatment. This raises the possibility that other cell types, e.g. lymphocytes and macrophages, contribute to tissue inflammation and destruction. Alexandre T. Rotta, M.D. Mauro Grossi, M.D. John E. Fisher, M.D. Howard Faden, M.D. Divisions of Pediatric Critical Care (ATR), Hematology and Oncology (MG) and Infectious Diseases (HF); Department of Pediatrics Department of Pathology (JEF) The Children's Hospital of Buffalo and State University of New York School of Medicine Buffalo, NY" @default.
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• W2008015488 title "DELAYED MYONECROSIS IN A LEUKEMIC PATIENT WITH INVASIVE GROUP A STREPTOCOCCAL DISEASE" @default.
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