- Email: [email protected]
Necrotizing Lip Infection Causing Septic Thrombophlebitis of the Neck: A Rare Variant of Lemierre Syndrome
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56
CASE REPORT
Q1 Q11
Q2
Necrotizing Lip Infection Causing Septic Thrombophlebitis of the Neck: A Rare Variant of Lemierre Syndrome Karl Cuddy, MD, DDS, MSc,* Nariman Saadat, MD, DDS,y Baber Khatib, MD, DDS,z and Ashish Patel, MD, DDSx Lemierre syndrome is an uncommon condition in which internal jugular vein thrombosis presents after recent oropharyngeal infection. Frequently, this is accompanied by septic emboli. This report outlines a variant of this disease process, with septic thrombophlebitis of the neck associated with a necrotizing skin infection of the lower lip and chin. A 25-year-old man with lower lip and chin swelling, initially managed with intravenous antibiotics, progressed to the development of a left facial vein thrombus, septic emboli to the lungs, and a necrotizing lower lip and chin infection that was managed with debridement, thrombectomy, and prolonged hemodynamic and pulmonary support. A necrotizing skin infection with thrombus of the jugular system and septic emboli is a very rare variant of Lemierre syndrome. Early recognition of an infection with septic emboli and/or necrotizing pathobiological findings allows for prompt antibiotic and surgical therapy, minimizing the mortality of these potentially lethal infections. Ó 2017 Published by Elsevier Inc on behalf of the American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg -:1-6, 2017 Septic thrombophlebitis of the head and neck is rare and most commonly associated with oropharyngeal infections or pharyngitis in which Fusobacterium necrophorum is the most frequently identified pathogen.1-5 Lemierre syndrome is classically defined as thrombosis of the internal jugular vein or one of its tributaries in a patient with an oropharyngeal source of infection.2 Septic thrombophlebitis from other head and neck sources including odontogenic, periorbital, and skin infections has been reported but is exceedingly rare.2-6 In 1936 Lemierre1 reported a 90% mortality rate in patients with this syndrome, although contemporary literature has reported a 5% mortality rate among patients treated for Lemierre syndrome.2 We present a case of a Staphylococcus aureus
necrotizing skin infection of the lower lip with common facial vein thrombosis and septic emboli to the lungs.
Case Report A 25-year-old man with an unremarkable medical history presented to Legacy Emanuel Medical Center with a 4-day period of progressively worsening lower lip and chin swelling. The oral and maxillofacial surgery department was consulted for assessment and management. The findings of the initial assessment were remarkable for tachycardia and a cellulitic process of the lower lip and chin without evidence of fluctuance, crepitus, or tissue necrosis. Low-grade Q3
*Fellow, Advanced Craniomaxillofacial Surgery and Trauma,
Address correspondence and reprint requests to Dr Cuddy:
Legacy Emanuel Medical Center, The Head and Neck Institute,
Legacy Emanuel Medical Center, The Head and Neck Institute,
Portland, OR.
1849 NW Kearney St, Ste 300, Portland, OR 97209; e-mail:
yResident, Division of Oral and Maxillofacial Surgery, Oregon
[email protected]
Health Science University, Portland, OR.
Received April 15 2017
zFellow, Advanced Craniomaxillofacial Surgery and Trauma,
Accepted May 26 2017 Ó 2017 Published by Elsevier Inc on behalf of the American Association of Oral
Legacy Emanuel Medical Center, The Head and Neck Institute, Portland, OR.
and Maxillofacial Surgeons
xHead and Neck Surgeon and Oral and Maxillofacial Surgeon,
0278-2391/17/30607-9
Legacy Emanuel Medical Center, The Head and Neck Institute,
http://dx.doi.org/10.1016/j.joms.2017.05.030
Portland, OR. Conflict of Interest Disclosures: None of the authors have any relevant financial relationship(s) with a commercial interest.
1 CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
57 58 59 60 61 62 63 64 65 66 67 68 69 70 71 72 73 74 75 76 77 78 79 80 81 82 83 84 85 86 87 88 89 90 91 92 93 94 95 96 97 98 99 100 101 102 103 104 105 106 107 108 109 110 111 112
113 114 115 116 117 118 119 120 121 122 123 124 125 126 127 128 129 130 131 132 133 134 135 136 137 138 139 140 141 142 143 144 145 146 147 148 149 150 151 152 153 154 155 156 157 158 159 160 161 162 163 164 165 166 167 168
2
Q4
Q5
Q6
SEPTIC THROMBOPHLEBITIS FROM NECROTIZING LIP INFECTION
left-sided chest pain was present on inspiration. The initial laboratory findings were remarkable for leukocytosis with marked bandemia, hyponatremia, and mild transaminitis. Diagnostic imaging included contrast-enhanced computed tomography (CT) of the face, which showed evidence of edema of the lower lip and chin without gas formation or fluid collection, as well as a nonocclusive thrombus of the left common facial vein. The chest radiograph findings were notable for bilateral scattered infiltrates (Fig 1). Subsequently, a diagnosis of lower lip and chin cellulitis with thrombophlebitis of the left common facial vein was made (Figs 2, 3). The patient was admitted, and antimicrobial therapy with intravenous (IV) ampicillin-sulbactam and vancomycin was initiated. On post-admission day 1, worsening tachycardia developed (heart rate >150 beats/min) and the patient required supplemental oxygen. The clinical examination findings were notable for worsening left-sided lower lip and chin fullness with concern for abscess development. In addition, substantial bilateral chest pain was present on deep inspiration. A CT angiogram of the chest was obtained and showed numerous lung nodules and small bilateral pleural effusions (Fig 4). The
internal medicine department was consulted; we initiated a therapeutic heparin infusion, and the patient underwent bedside incision and drainage of the lower lip and chin under local anesthesia. At this time, the plan included a trial of medical management with a plan to pursue further surgical care if the patient did not improve over the next 12 to 24 hours. On post-admission day 3, the patient continued to have sepsis with worsening fever. He was subse- Q7 quently taken to the operating room for ligation and resection of the left common facial vein and thrombus, as well as repeat incision and drainage with debridement of the lower lip and chin (Figs 5-7). The intraoperative course was uneventful. While the patient was in the postanesthesia care unit, we noted the development of worsening tachycardia, tachypnea, hypotension, and interval drastic worsening of lower lip and chin edema with an area of necrosis measuring 1 cm2. The patient was immediately taken back to the operating room for emergent intubation, resuscitation, and further debridement of the lower lip necrosis. Postoperatively, he remained intubated and was transferred to the intensive care unit.
FIGURE 1. Chest radiograph on admission showing bilateral scattered infiltrates. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
Q10
169 170 171 172 173 174 175 176 177 178 179 180 181 182 183 184 185 186 187 188 189 190 191 192 193 194 195 196 197 198 199 200 201 202 203 204 205 206 207 208 209 210 211 212 213 214 215 216 217 218 219 220 221 222 223 224
3
CUDDY ET AL
225 226 227 228 229 230 231 232 233 234 235 236 237 238 239 240 241 242 243 244 245 246 247 248 249 250 251 252 253 254 255 256 257 258 259 260 261 262 263 264 265 266 267 268 269 270 271 272 273 274 275 276 277 278 279 280
FIGURE 2. Axial section of facial computed tomography scan on admission showing no abscess formation in region of lower lip. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
Q8
At this time, the patient’s problem list included a necrotizing soft tissue infection of the lower lip and chin with septic thrombophlebitis of the left common facial vein (Figs 8, 9), hypoxic respiratory failure, hyponatremia, transaminitis, and septic shock. On post-admission day 4, no further tissue necrosis was identified and the patient was weaned from hemodynamic support and extubated. During this process, the infectious disease service was consulted, and antimicrobial therapy was broadened to piperacillintazobactam and vancomycin. After culture and sensitivity analysis was completed, antimicrobial therapy was changed to nafcillin, clindamycin, and metronidazole. On post-admission day 5, the patient continued to have fever and tachypnea ranging from 30 to 40 respirations per minute. Subsequent CT of the neck and chest showed an interval increase in pleural fluid accumulation prompting chest tube placement. During the next several days, the patient’s respiratory status improved, the sepsis resolved, and the chest
tube was removed. The definitive culture from both Q9 the lower lip and facial vein grew methicillinsensitive S aureus; the patient was discharged on post-admission day 20 with a plan to receive IV nafcillin for 1 month.
Discussion Head and neck skin infections are relatively common, varying in severity, with treatment ranging from a short course of oral antibiotics on an outpatient basis to prolonged inpatient care with repeated debridement. The pathogenic organisms isolated are variable but frequently contain S aureus as an offending pathogen. Isolated cases of necrotizing neck infections with septic thrombophlebitis have been reported, but the combined presentation of these conditions is extremely rare.6 The initial clinical examination and CT scan in our patient did not show findings suggestive of a
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
281 282 283 284 285 286 287 288 289 290 291 292 293 294 295 296 297 298 299 300 301 302 303 304 305 306 307 308 309 310 311 312 313 314 315 316 317 318 319 320 321 322 323 324 325 326 327 328 329 330 331 332 333 334 335 336
337 338 339 340 341 342 343 344 345 346 347 348 349 350 351 352 353 354 355 356 357 358 359 360 361 362 363 364 365 366 367 368 369 370 371 372 373 374 375 376 377 378 379 380 381 382 383 384 385 386 387 388 389 390 391 392
4
SEPTIC THROMBOPHLEBITIS FROM NECROTIZING LIP INFECTION
FIGURE 3. Coronal section of neck computed tomography scan on admission showing presence of nonocclusive thrombus (arrow) of left common facial vein. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
necrotizing skin infection. Furthermore, the findings of assessment adjuncts such as the Laboratory Risk Indicator for Necrotizing Fasciitis7 and standardized
early warning system8 were not suggestive of a necrotizing skin infection. Within 24 to 36 hours of admission, the patient’s clinical status substantially
FIGURE 4. Axial image of upper chest from computed tomography scan obtained on hospital day 1 showing bilateral pulmonary nodules and small bilateral effusions. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
393 394 395 396 397 398 399 400 401 402 403 404 405 406 407 408 409 410 411 412 413 414 415 416 417 418 419 420 421 422 423 424 425 426 427 428 429 430 431 432 433 434 435 436 437 438 439 440 441 442 443 444 445 446 447 448
5
Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
FIGURE 7. Intraoperative photograph showing defect on left side of neck from thrombectomy. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
Appropriate empiric IV antibiotic therapy was administered at the time of the patient’s presentation to the emergency department and continued during the admission. Despite antibiotic therapy, the patient’s condition worsened and surgical debridement was planned. Although no clinical trials have supported routine thrombectomy in the management of Lemierre syndrome,2-4 we elected to proceed with limited neck exploration, left facial vein ligation, and thrombectomy, in addition to debridement of the lip and chin, because of the rapidly worsening clinical course with progression of emboli and subsequent pulmonary dysfunction requiring respiratory and hemodynamic support. Septic thrombophlebitis of the facial or jugular veins caused by skin infection is extremely rare. Our case appears to be the first documented case of septic
print & web 4C=FPO
deteriorated, with tachypnea, fever, hypoxia, and pleuritic chest pain in addition to pain at the primary site of infection (lip and chin) and neck. The combination of these findings with septic thrombus of the left common facial vein is consistent with an atypical presentation of Lemierre disease.
print & web 4C=FPO
FIGURE 5. Intraoperative photograph showing preoperative appearance of lower lip.
print & web 4C=FPO
449 450 451 452 453 454 455 456 457 458 459 460 461 462 463 464 465 466 467 468 469 470 471 472 473 474 475 476 477 478 479 480 481 482 483 484 485 486 487 488 489 490 491 492 493 494 495 496 497 498 499 500 501 502 503 504
print & web 4C=FPO
CUDDY ET AL
FIGURE 6. Intraoperative photograph showing limited dissection specimen from left side of neck including left common facial vein.
FIGURE 8. Hematoxylin-eosin–stained specimen (original magnification 50) showing intravascular thrombus.
Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
505 506 507 508 509 510 511 512 513 514 515 516 517 518 519 520 521 522 523 524 525 526 527 528 529 530 531 532 533 534 535 536 537 538 539 540 541 542 543 544 545 546 547 548 549 550 551 552 553 554 555 556 557 558 559 560
SEPTIC THROMBOPHLEBITIS FROM NECROTIZING LIP INFECTION
conjunction with antibiotic therapy. Thrombectomy may be considered in situations of rapidly worsening systemic illness in patients already undergoing antibiotic therapy. Awareness of rare variations in the presentation of septic thrombophlebitis of the neck, with early initiation of appropriate surgical and antimicrobial therapy, may help to minimize morbidity and mortality associated with this uncommon disease entity.
References
print & web 4C=FPO
561 562 563 564 565 566 567 568 569 570 571 572 573 574 575 576 577 578 579 580 581 582 583 584 585 586 587 588 589 590 591 592 593
6
FIGURE 9. Hematoxylin-eosin–stained specimen (original magnification 50) showing lip tissue with focal necrosis. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
thrombophlebitis of the facial vein from a necrotizing skin infection. Aggressive, early surgical management of necrotizing skin infections with frequent re-evaluation of the affected tissue is imperative, in
1. Lemierre A: On certain septicemias due to anaerobic organisms. Lancet 1:701, 1936 2. Karkos PD, Asrani S, Karkos CD, et al: Lemierre’s syndrome: A systematic review. Laryngoscope 119:1552, 2009 3. Noy D, Rachmiel A, Levy-Faber D, Emodi O: Lemierre’s syndrome from odontogenic infection: Review of the literature and case description. Ann Maxillofac Surg 5:219, 2015 4. Righini CA, Karkas A, Tourniaire R, et al: Lemierre syndrome: Study of 11 cases and literature review. Head Neck 36:1044, 2014 5. Yoshikawa H, Suzuki M, Nemoto N, et al: Internal jugular thrombophlebitis caused by dermal infection. Intern Med 50:447, 2011 6. Deganello A, Gallo O, Gitti G, De Campora E: Necrotizing fasciitis of the neck associated with Lemierre syndrome. Acta Otorhinolaryngol Ital 29:160, 2009 7. Wong CH, Khin LW, Heng KS, et al: The LRINEC (Laboratory Risk Indicator for Necrotizing Fasciitis) score: A tool for distinguishing necrotizing fasciitis from other soft tissue infections. Crit Care Med 32:1535, 2004 8. Marwick C, Broomhall J, McCowan C, et al: Severity assessment of skin and soft tissue infections: Cohort study of management and outcomes for hospitalized patients. J Antimicrob Chemother 66: 387, 2011
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
594 595 596 597 598 599 600 601 602 603 604 605 606 607 608 609 610 611 612 613 614 615 616 617 618 619 620 621 622 623 624 625 626
CASE REPORT
Q1 Q11
Q2
Necrotizing Lip Infection Causing Septic Thrombophlebitis of the Neck: A Rare Variant of Lemierre Syndrome Karl Cuddy, MD, DDS, MSc,* Nariman Saadat, MD, DDS,y Baber Khatib, MD, DDS,z and Ashish Patel, MD, DDSx Lemierre syndrome is an uncommon condition in which internal jugular vein thrombosis presents after recent oropharyngeal infection. Frequently, this is accompanied by septic emboli. This report outlines a variant of this disease process, with septic thrombophlebitis of the neck associated with a necrotizing skin infection of the lower lip and chin. A 25-year-old man with lower lip and chin swelling, initially managed with intravenous antibiotics, progressed to the development of a left facial vein thrombus, septic emboli to the lungs, and a necrotizing lower lip and chin infection that was managed with debridement, thrombectomy, and prolonged hemodynamic and pulmonary support. A necrotizing skin infection with thrombus of the jugular system and septic emboli is a very rare variant of Lemierre syndrome. Early recognition of an infection with septic emboli and/or necrotizing pathobiological findings allows for prompt antibiotic and surgical therapy, minimizing the mortality of these potentially lethal infections. Ó 2017 Published by Elsevier Inc on behalf of the American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg -:1-6, 2017 Septic thrombophlebitis of the head and neck is rare and most commonly associated with oropharyngeal infections or pharyngitis in which Fusobacterium necrophorum is the most frequently identified pathogen.1-5 Lemierre syndrome is classically defined as thrombosis of the internal jugular vein or one of its tributaries in a patient with an oropharyngeal source of infection.2 Septic thrombophlebitis from other head and neck sources including odontogenic, periorbital, and skin infections has been reported but is exceedingly rare.2-6 In 1936 Lemierre1 reported a 90% mortality rate in patients with this syndrome, although contemporary literature has reported a 5% mortality rate among patients treated for Lemierre syndrome.2 We present a case of a Staphylococcus aureus
necrotizing skin infection of the lower lip with common facial vein thrombosis and septic emboli to the lungs.
Case Report A 25-year-old man with an unremarkable medical history presented to Legacy Emanuel Medical Center with a 4-day period of progressively worsening lower lip and chin swelling. The oral and maxillofacial surgery department was consulted for assessment and management. The findings of the initial assessment were remarkable for tachycardia and a cellulitic process of the lower lip and chin without evidence of fluctuance, crepitus, or tissue necrosis. Low-grade Q3
*Fellow, Advanced Craniomaxillofacial Surgery and Trauma,
Address correspondence and reprint requests to Dr Cuddy:
Legacy Emanuel Medical Center, The Head and Neck Institute,
Legacy Emanuel Medical Center, The Head and Neck Institute,
Portland, OR.
1849 NW Kearney St, Ste 300, Portland, OR 97209; e-mail:
yResident, Division of Oral and Maxillofacial Surgery, Oregon
[email protected]
Health Science University, Portland, OR.
Received April 15 2017
zFellow, Advanced Craniomaxillofacial Surgery and Trauma,
Accepted May 26 2017 Ó 2017 Published by Elsevier Inc on behalf of the American Association of Oral
Legacy Emanuel Medical Center, The Head and Neck Institute, Portland, OR.
and Maxillofacial Surgeons
xHead and Neck Surgeon and Oral and Maxillofacial Surgeon,
0278-2391/17/30607-9
Legacy Emanuel Medical Center, The Head and Neck Institute,
http://dx.doi.org/10.1016/j.joms.2017.05.030
Portland, OR. Conflict of Interest Disclosures: None of the authors have any relevant financial relationship(s) with a commercial interest.
1 CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
57 58 59 60 61 62 63 64 65 66 67 68 69 70 71 72 73 74 75 76 77 78 79 80 81 82 83 84 85 86 87 88 89 90 91 92 93 94 95 96 97 98 99 100 101 102 103 104 105 106 107 108 109 110 111 112
113 114 115 116 117 118 119 120 121 122 123 124 125 126 127 128 129 130 131 132 133 134 135 136 137 138 139 140 141 142 143 144 145 146 147 148 149 150 151 152 153 154 155 156 157 158 159 160 161 162 163 164 165 166 167 168
2
Q4
Q5
Q6
SEPTIC THROMBOPHLEBITIS FROM NECROTIZING LIP INFECTION
left-sided chest pain was present on inspiration. The initial laboratory findings were remarkable for leukocytosis with marked bandemia, hyponatremia, and mild transaminitis. Diagnostic imaging included contrast-enhanced computed tomography (CT) of the face, which showed evidence of edema of the lower lip and chin without gas formation or fluid collection, as well as a nonocclusive thrombus of the left common facial vein. The chest radiograph findings were notable for bilateral scattered infiltrates (Fig 1). Subsequently, a diagnosis of lower lip and chin cellulitis with thrombophlebitis of the left common facial vein was made (Figs 2, 3). The patient was admitted, and antimicrobial therapy with intravenous (IV) ampicillin-sulbactam and vancomycin was initiated. On post-admission day 1, worsening tachycardia developed (heart rate >150 beats/min) and the patient required supplemental oxygen. The clinical examination findings were notable for worsening left-sided lower lip and chin fullness with concern for abscess development. In addition, substantial bilateral chest pain was present on deep inspiration. A CT angiogram of the chest was obtained and showed numerous lung nodules and small bilateral pleural effusions (Fig 4). The
internal medicine department was consulted; we initiated a therapeutic heparin infusion, and the patient underwent bedside incision and drainage of the lower lip and chin under local anesthesia. At this time, the plan included a trial of medical management with a plan to pursue further surgical care if the patient did not improve over the next 12 to 24 hours. On post-admission day 3, the patient continued to have sepsis with worsening fever. He was subse- Q7 quently taken to the operating room for ligation and resection of the left common facial vein and thrombus, as well as repeat incision and drainage with debridement of the lower lip and chin (Figs 5-7). The intraoperative course was uneventful. While the patient was in the postanesthesia care unit, we noted the development of worsening tachycardia, tachypnea, hypotension, and interval drastic worsening of lower lip and chin edema with an area of necrosis measuring 1 cm2. The patient was immediately taken back to the operating room for emergent intubation, resuscitation, and further debridement of the lower lip necrosis. Postoperatively, he remained intubated and was transferred to the intensive care unit.
FIGURE 1. Chest radiograph on admission showing bilateral scattered infiltrates. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
Q10
169 170 171 172 173 174 175 176 177 178 179 180 181 182 183 184 185 186 187 188 189 190 191 192 193 194 195 196 197 198 199 200 201 202 203 204 205 206 207 208 209 210 211 212 213 214 215 216 217 218 219 220 221 222 223 224
3
CUDDY ET AL
225 226 227 228 229 230 231 232 233 234 235 236 237 238 239 240 241 242 243 244 245 246 247 248 249 250 251 252 253 254 255 256 257 258 259 260 261 262 263 264 265 266 267 268 269 270 271 272 273 274 275 276 277 278 279 280
FIGURE 2. Axial section of facial computed tomography scan on admission showing no abscess formation in region of lower lip. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
Q8
At this time, the patient’s problem list included a necrotizing soft tissue infection of the lower lip and chin with septic thrombophlebitis of the left common facial vein (Figs 8, 9), hypoxic respiratory failure, hyponatremia, transaminitis, and septic shock. On post-admission day 4, no further tissue necrosis was identified and the patient was weaned from hemodynamic support and extubated. During this process, the infectious disease service was consulted, and antimicrobial therapy was broadened to piperacillintazobactam and vancomycin. After culture and sensitivity analysis was completed, antimicrobial therapy was changed to nafcillin, clindamycin, and metronidazole. On post-admission day 5, the patient continued to have fever and tachypnea ranging from 30 to 40 respirations per minute. Subsequent CT of the neck and chest showed an interval increase in pleural fluid accumulation prompting chest tube placement. During the next several days, the patient’s respiratory status improved, the sepsis resolved, and the chest
tube was removed. The definitive culture from both Q9 the lower lip and facial vein grew methicillinsensitive S aureus; the patient was discharged on post-admission day 20 with a plan to receive IV nafcillin for 1 month.
Discussion Head and neck skin infections are relatively common, varying in severity, with treatment ranging from a short course of oral antibiotics on an outpatient basis to prolonged inpatient care with repeated debridement. The pathogenic organisms isolated are variable but frequently contain S aureus as an offending pathogen. Isolated cases of necrotizing neck infections with septic thrombophlebitis have been reported, but the combined presentation of these conditions is extremely rare.6 The initial clinical examination and CT scan in our patient did not show findings suggestive of a
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
281 282 283 284 285 286 287 288 289 290 291 292 293 294 295 296 297 298 299 300 301 302 303 304 305 306 307 308 309 310 311 312 313 314 315 316 317 318 319 320 321 322 323 324 325 326 327 328 329 330 331 332 333 334 335 336
337 338 339 340 341 342 343 344 345 346 347 348 349 350 351 352 353 354 355 356 357 358 359 360 361 362 363 364 365 366 367 368 369 370 371 372 373 374 375 376 377 378 379 380 381 382 383 384 385 386 387 388 389 390 391 392
4
SEPTIC THROMBOPHLEBITIS FROM NECROTIZING LIP INFECTION
FIGURE 3. Coronal section of neck computed tomography scan on admission showing presence of nonocclusive thrombus (arrow) of left common facial vein. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
necrotizing skin infection. Furthermore, the findings of assessment adjuncts such as the Laboratory Risk Indicator for Necrotizing Fasciitis7 and standardized
early warning system8 were not suggestive of a necrotizing skin infection. Within 24 to 36 hours of admission, the patient’s clinical status substantially
FIGURE 4. Axial image of upper chest from computed tomography scan obtained on hospital day 1 showing bilateral pulmonary nodules and small bilateral effusions. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
393 394 395 396 397 398 399 400 401 402 403 404 405 406 407 408 409 410 411 412 413 414 415 416 417 418 419 420 421 422 423 424 425 426 427 428 429 430 431 432 433 434 435 436 437 438 439 440 441 442 443 444 445 446 447 448
5
Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
FIGURE 7. Intraoperative photograph showing defect on left side of neck from thrombectomy. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
Appropriate empiric IV antibiotic therapy was administered at the time of the patient’s presentation to the emergency department and continued during the admission. Despite antibiotic therapy, the patient’s condition worsened and surgical debridement was planned. Although no clinical trials have supported routine thrombectomy in the management of Lemierre syndrome,2-4 we elected to proceed with limited neck exploration, left facial vein ligation, and thrombectomy, in addition to debridement of the lip and chin, because of the rapidly worsening clinical course with progression of emboli and subsequent pulmonary dysfunction requiring respiratory and hemodynamic support. Septic thrombophlebitis of the facial or jugular veins caused by skin infection is extremely rare. Our case appears to be the first documented case of septic
print & web 4C=FPO
deteriorated, with tachypnea, fever, hypoxia, and pleuritic chest pain in addition to pain at the primary site of infection (lip and chin) and neck. The combination of these findings with septic thrombus of the left common facial vein is consistent with an atypical presentation of Lemierre disease.
print & web 4C=FPO
FIGURE 5. Intraoperative photograph showing preoperative appearance of lower lip.
print & web 4C=FPO
449 450 451 452 453 454 455 456 457 458 459 460 461 462 463 464 465 466 467 468 469 470 471 472 473 474 475 476 477 478 479 480 481 482 483 484 485 486 487 488 489 490 491 492 493 494 495 496 497 498 499 500 501 502 503 504
print & web 4C=FPO
CUDDY ET AL
FIGURE 6. Intraoperative photograph showing limited dissection specimen from left side of neck including left common facial vein.
FIGURE 8. Hematoxylin-eosin–stained specimen (original magnification 50) showing intravascular thrombus.
Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
505 506 507 508 509 510 511 512 513 514 515 516 517 518 519 520 521 522 523 524 525 526 527 528 529 530 531 532 533 534 535 536 537 538 539 540 541 542 543 544 545 546 547 548 549 550 551 552 553 554 555 556 557 558 559 560
SEPTIC THROMBOPHLEBITIS FROM NECROTIZING LIP INFECTION
conjunction with antibiotic therapy. Thrombectomy may be considered in situations of rapidly worsening systemic illness in patients already undergoing antibiotic therapy. Awareness of rare variations in the presentation of septic thrombophlebitis of the neck, with early initiation of appropriate surgical and antimicrobial therapy, may help to minimize morbidity and mortality associated with this uncommon disease entity.
References
print & web 4C=FPO
561 562 563 564 565 566 567 568 569 570 571 572 573 574 575 576 577 578 579 580 581 582 583 584 585 586 587 588 589 590 591 592 593
6
FIGURE 9. Hematoxylin-eosin–stained specimen (original magnification 50) showing lip tissue with focal necrosis. Cuddy et al. Septic Thrombophlebitis From Necrotizing Lip Infection. J Oral Maxillofac Surg 2017.
thrombophlebitis of the facial vein from a necrotizing skin infection. Aggressive, early surgical management of necrotizing skin infections with frequent re-evaluation of the affected tissue is imperative, in
1. Lemierre A: On certain septicemias due to anaerobic organisms. Lancet 1:701, 1936 2. Karkos PD, Asrani S, Karkos CD, et al: Lemierre’s syndrome: A systematic review. Laryngoscope 119:1552, 2009 3. Noy D, Rachmiel A, Levy-Faber D, Emodi O: Lemierre’s syndrome from odontogenic infection: Review of the literature and case description. Ann Maxillofac Surg 5:219, 2015 4. Righini CA, Karkas A, Tourniaire R, et al: Lemierre syndrome: Study of 11 cases and literature review. Head Neck 36:1044, 2014 5. Yoshikawa H, Suzuki M, Nemoto N, et al: Internal jugular thrombophlebitis caused by dermal infection. Intern Med 50:447, 2011 6. Deganello A, Gallo O, Gitti G, De Campora E: Necrotizing fasciitis of the neck associated with Lemierre syndrome. Acta Otorhinolaryngol Ital 29:160, 2009 7. Wong CH, Khin LW, Heng KS, et al: The LRINEC (Laboratory Risk Indicator for Necrotizing Fasciitis) score: A tool for distinguishing necrotizing fasciitis from other soft tissue infections. Crit Care Med 32:1535, 2004 8. Marwick C, Broomhall J, McCowan C, et al: Severity assessment of skin and soft tissue infections: Cohort study of management and outcomes for hospitalized patients. J Antimicrob Chemother 66: 387, 2011
CRP 5.4.0 DTD YJOMS57827_proof 22 June 2017 6:43 pm CE KO
594 595 596 597 598 599 600 601 602 603 604 605 606 607 608 609 610 611 612 613 614 615 616 617 618 619 620 621 622 623 624 625 626