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staining with a fluorescein-conjugated species-spe-cific monoclonal antibody, because this method is specific.6_{This recommendation came out of an}

inves-tigation of an “outbreak” of suspected sexual abuse in an institution for the mentally disabled in Ohio that occurred in 1990.15_{As in the present case, a large}

commercial laboratory was using an EIA for culture confirmation. All the positive specimens, which were rectal specimens, were subsequently proved to be false-positives. The use of an EIA for culture confir-mation has the potential to generate false-positive results because the EIA detects solubilized chlamyd-ial antigens that would be derived from the inocu-lated cell culture as well as cross-reacting antigens from other organisms present in clinical specimens. This is more likely to happen with rectal specimens as the number of potentially cross-reacting bacteria present are high and may be amplified in the cell culture system. This is probably what happened in case 4, as the positive cultures that were confirmed by EIA could not be confirmed by FA staining and should have been reported as negative.

At the least the use of these inappropriate tests resulted in unnecessary retesting and at the worst, resulted in unnecessary hospitalization, erroneous reports of sexual abuse and possibly unjustified prosecution and incarceration. The current recom-mendations for testing forC trachomatisin the setting of suspected child sexual abuse were first made in 1989 and reiterated in the 1993 and 1998 CDC Sexu-ally Transmitted Disease Guidelines.4 – 6 _{Because of the}

social and legal implications it is important that prac-titioners be aware of these recommendations and require that commercial laboratories adhere to ap-provedC trachomatisculture methods.

Margaret R. Hammerschlag, MD Stephen Ajl, MD

Departments of Pediatrics and Medicine

State University of New York Health Sciences Center at Brooklyn

Brooklyn, NY 11203-2098

Danielle Laraque, MD

Department of Pediatrics

College of Physicians and Surgeons

Columbia University at Harlem Hospital Center New York, NY 10037

REFERENCES

1. Hammerschlag MR, Rettig PJ, Shields ME. False positive results with the use of chlamydial antigen detection tests in the evaluation of sus-pected sexual abuse in children.Pediatr Infect Dis J. 1988;7:11–14 2. Porder K, Sanchez N, Roblin PM, McHugh M, Hammerschlag MR. Lack

of specificity of chlamydiazyme for detection of vaginal chlamydial infection in prepubertal girls.Pediatr Infect Dis J. 1989;8:358 –360 3. Hauger SB, Brown J, Agre F, Sahraie F, Ortiz R, Ellner P. Failure to

direct fluorescent antibody staining to detectChlamydia trachomatisfrom genital tract sites of prepubertal children at risk for sexual abuse.Pediatr Infect Dis J. 1988;7:660 – 661

4. Centers for Disease Control. Sexually Transmitted Diseases Treatment Guidelines.MMWR Morbid Mortal Wkly Rep.1989;38:S-8

5. Centers for Disease Control. Sexually Transmitted Diseases Treatment Guidelines.MMWR Morbid Mortal Wkly Rep.1993;42:RR-14

6. Centers for Disease Control. 1998 Guidelines for Treatment of Sexually Transmitted Diseases.MMWR Morbid Mortal Wkly Rep. 1998;47:RR-1 7. Riordan T Ellis DA, Mathews PI, Ratcliffe SF. False positive results with

an ELISA for detection of chlamydia antigen.J Clin Pathol. 1986;39: 1276 –1277

8. Goudswaard F, Sabbe L, van Belzen C. Interference by gram-negative bacteria in the enzyme immuno-assay for detectingChlamydia trachoma-tis. J Infect Dis. 1989;18:94 –96

9. Pate MS, Hook EW. Laboratory to laboratory variation inChlamydia trachomatisculture practices.Sex Transm Dis. 1995;22:322–326 10. Bell TA, Stamm WE, Wang SP et al. Chronic Chlamydia trachomatis

infections in infants.JAMA. 1992;267:400 – 402

11. Everett VD, Ingram DL, Flick LAR, Russell TA, Tropez-Sims ST, Mc-Fadden AY. A comparison of sexually transmitted diseases (STDs) found in a total of 696 boys and 2973 girls evaluated for sexual abuse. Pediatr Res. 1998;43:91. Abstract

12. Black CM. Current methods of laboratory diagnosis ofChlamydia tra-chomatisinfections.Clin Microbiol Rev. 1997;10:160 –184

13. Schachter J. Immunodiagnosis of sexually transmitted disease. Yale J Biol Med. 1985;58:443– 452

14. Mathews-Greer J, Sloop G, Springer A, McRae K, LaHaye E, Jamison R. Comparison of detection methods forChlamydia trachomatisin speci-mens obtained from pediatric victims of suspected sexual abuse.Pediatr Infect Dis J. 1999;18:165–167

15. Centers for Disease Control. False-positive results with the use of chla-mydia tests in the evaluation of suspected sexual abuse—Ohio, 1990. MMWR Morbid Mortal Wkly Rep. 1991;39:932–935

Spinal Epidural Abscess in

Preverbal Children: A Case Report

With Currarino Triad

ABSTRACT. Spinal epidural abscess is rare in prever-bal children and leads to permanent neurologic deficits if not treated promptly. Currarino triad (anorectal malfor-mation, sacral bony abnormality and presacral mass) is also rare in children. We report the association of exten-sive spinal epidural abscess and Currarino triad in a young child. Pediatrics 1999;104:1139 –1142; Currarino triad, spinal epidural abscess, pediatrics.

ABBREVIATIONS. LP, lumbar puncture; MRI, magnetic reso-nance imaging; POD, postoperative day; SEA, spinal epidural abscess; CSF, cerebrospinal fluid.

CASE REPORT

A 5-week-old white boy presented to the emergency depart-ment with fever, irritability, and grunting for 1 day. Additionally, the infant seemed to cry and strain with every stool that consisted of small smears since he was 2 weeks old. Mylicon (J & J Merck, Fort Washington, PA) drops, glycerin suppositories, and multiple formula changes did not alter this. The infant was feeding and growing well and had no ill contacts. He was delivered full-term and birth history was remarkable for maternal cervical carriage of group Bb-hemolyticStreptococcustreated with antibiotics before delivery. At rupture of membranes approximately 14 hours before delivery, particulate meconium was noted in the amniotic fluid, which cleared after amnioinfusion. The postnatal course was oth-erwise uneventful.

In the emergency department, the infant appeared well nour-ished, vigorous, and irritable but easily consolable. Skin was well perfused and temperature was 38.7°C, pulse was 182/minute, respiratory rate was 52/minute, and blood pressure was 85/57 mm Hg. Anterior fontanelle was soft and flat and the neck was supple. Abdomen was nontender, nondistended, and without any

Received for publication Feb 22, 1999; accepted May 19, 1999.

Reprint requests to (J.D.L.) Department of Pediatrics, Division of Emer-gency Medicine, Washington University School of Medicine, St Louis Chil-dren’s Hospital, One ChilChil-dren’s Place, St Louis, MO 63110-1077. E-mail: [email protected]

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palpable mass. There was a 1-cm sclerosed umbilical stump present, without any drainage, odor or periumbilical erythema, or induration. The anus appeared normally positioned with an intact anal wink. Spine was straight with no tenderness to palpation and no overlying erythema or induration. Tone, reflexes, and strength were intact and he moved all extremities well.

Blood, urine and stool cultures were obtained. Lumbar punc-ture (LP) was performed through the L4-L5 interspace and thick pus flowed through the needle on advancing it a small distance beyond the skin. Although there was no overlying skin erythema, induration, palpable mass, or fluctuance, the possibility of an abscess was considered. A second LP was attempted at the L2-L3 interspace and again thick pus flowed through the spinal needle. Both were sent for Gram stain and culture.

Laboratory studies revealed a white blood cell count of 21 700 cells/mL with 9 bands, 54 segmented neutrophils, 25 lymphocytes, 10 mononucleocytes, and 2 immature polys; hemoglobin 8.1mg/ dL, platelets 575 000. Chest radiograph, urinalysis, and serum electrolytes were normal. Gram stain of the fluid obtained from the LP showed Gram-positive cocci in clusters, Gram-negative rods, and Gram-positive cocci in pairs. The Gram stain on the second specimen showed Gram-positive cocci in clusters and Gram-negative rods. Vancomycin, meropenem, and gentamicin were started and a head and spinal magnetic resonance imaging (MRI) was done to evaluate the possibility of an epidural abscess. The MRI of the head was normal but the spine showed a large epidural collection extend-ing from C3 to the sacrum compressextend-ing the thecal sac anteriorly and communicating with a large, predominantly cystic presacral mass of the same imaging characteristics as the epidural collection, consistent with an extensive epidural abscess.

The infant was taken emergently to the operating room by the pediatric neurosurgeons. A small incision was made posteriorly just below the coccyx and after dissection through the subcutane-ous tissue, a large abscess cavity anterior to the sacrum containing green purulent fluid was encountered, irrigated, and packed. A posterior laminectomy was done at the L2-L3 level and eruption of greenish purulent fluid was again noted. A ventricular catheter was used to drain the epidural abscess by threading it up the epidural space rostrally to the cervical region with irrigation along the way until slow return of clear fluid, and threading it down caudally in the same manner. This fluid was also sent for Gram stain and culture. Although the presacral abscess cavity commu-nicated with the epidural abscess, there was no communication of that cavity with bowel or other adjacent abdominal structures.

The infant was taken postoperatively to the pediatric intensive care unit for management of hypotension, decreased perfusion, and oliguria that responded to intravenous fluids and pressor support. He was extubated by postoperative day (POD) 1. The Gram stain from the operating room specimens from the abscess cavity and epidural space showed Gram-positive cocci in clusters and Gram-negative rods. The cultures obtained during the initial LP at L4 –5and L2–3grewS aureus,Enterococcus, Bacteroides fragilis,

andPeptococcus magnus. On POD 2, the infant became afebrile and feedings were initiated. Anal dilatation was performed twice daily for mild anal stenosis. MRI of the spine done on PODs 2 and 9 showed no residual fluid collection in the presacral and epidural space. Initial blood, urine, stool cultures, sweat chloride and ni-troblue tetrazolium tests were negative. The patient underwent a hypaque enema and no fistula between the colon and the presacral space or spinal canal was visualized. However, an anomalous lower sacrum with a sickle-like configuration and partial absence of the lower right segments was noted. This was suggestive of Currarino triad: a sacral defect, anorectal malformation, and pre-sacral mass.

He was discharged from the hospital on day 10 to complete 4 weeks of antibiotic therapy with vancomycin and meropenam. The infant did well until 6 months of age when he presented with a 1-week history of straining to defecate and clay-like stool smears. A contrast enema revealed an anteriorly displaced rectum and demonstrated the sickle sacral anomaly. CT showed a presa-cral mass, a portion of which was fatty density abutting the defect in the bony sacrum. Subsequently, exploratory laparotomy re-vealed a retrorectal, presacral mass containing pus that grew

PseudomonasandCitrobacter. The mass was removed and identi-fied as a benign dermoid cyst.

DISCUSSION

Spinal epidural abscess (SEA) is uncommon in children. Although the incidence is unknown, .60 cases have been reported in children. Walter et al1

reported the largest series of SEA in 12 preverbal children. Six other cases of SEA in preverbal children including ours have been reported.2–5

Our case is the only case of SEA associated with the Currarino triad reported in the literature. The Currarino triad was described by Currarino in 19816

and is defined by the triad of an anorectal malfor-mation, a sacrococcygeal bony abnormality, and a presacral mass. More than 200 cases have been re-ported in the literature.7 _{The pathogenesis of the}

syndrome is postulated to be the result of abnormal separation of the neuroectoderm from the endoderm early on in fetal life.8 _{Many cases are familial, and}

family screening often uncovers 1 or 2 features of the syndrome in relatives.7_{Both an autosomal dominant}

and x-linked dominant pattern has been postulated, although the exact genetic defect remains unclear.7

The anorectal malformation is most frequently ano-rectal stenosis8_{and can range from rectal stenosis to}

anal ectopia and atresia. The bony defect is com-monly a sickle or Scimitar sacrum8 _{but can range}

from coccygeal lateral deviation to absence of distal sacral segments.7 _{The presacral mass is most}

fre-quently an anterior meningomyelocele (68% in one series), followed by benign teratoma (18%); malig-nancy is rare (1%).7_{The most common presentation}

is chronic constipation; in fact, one author9_suggests

that cases presenting with chronic constipation, a sacral bony defect, and a presacral mass should be considered Currarino syndrome, even in the absence of measurable anorectal stenosis. Kochling et al7_lists

meningitis as a dangerous complication of cases with an anal or rectal fistula communicating with the spinal canal, and meningitis associated with the Cur-rarino triad has been reported.8_{Our case is the only}

reported case of Currarino syndrome with an infec-tion of the presacral mass spreading to the epidural space. No communication between the presacral ab-scess and the spinal canal was grossly visualized either operative procedures. The chronic constipa-tion causing frequent episodes of elevated intraab-dominal pressure may have caused preferential shunting of venous blood to Batson’s plexus, predis-posing to hematogenous seeding of the epidural space from the presacral abscess. However, the exact mechanism of development of SEA in our patient remains uncertain.

Early recognition of SEA is of paramount impor-tance, as misdiagnosis or delay in treatment can lead to catastrophic sequelae, including sepsis, spinal de-formity, permanent neurologic deficit, and death. The diagnosis may be especially challenging in pre-verbal children because the initial signs and symp-toms are subtle and nonspecific: fever, irritability, eme-sis, crying with handling or movement, decreased appetite, and upper respiratory tract symptoms. Focal findings such as tenderness over the involved vertebral level are often absent.1,10,11_{Because of this, the diagnosis}

of SEA in infants is often delayed until the onset of

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neurologic deficits. Indeed, in all 6 infant cases re-viewed by Enberg and Kaplan11_{and 11 out of 12 cases}

reviewed by Walter et al,1_{the diagnosis of SEA was}

delayed until evidence of spinal cord impairment was apparent. Most of these infants had permanent neuro-logic injury and poor outcomes.

SEA in children occurs primarily by hematoge-nous spread of the causative organism from distant sites.1,10,11_{Many authors point to the spinal epidural}

network of veins, Batson’s plexus, as a route of in-fection.10 –12_{This plexus consists of many veins in the}

epidural space that anastomose at each spinal seg-ment with the veins of the thoracic and abdominal cavities.13 _{It is thought that venous blood flowing}

from the lower half of the body to the inferior vena cava may be shunted preferentially to this low pres-sure venous system when the intraabdominal or in-trapelvic pressure is elevated,10 –12_thereby

predispos-ing the epidural space to seed durpredispos-ing bacteremia. Local trauma appears to be a predisposing factor to SEA in older children.1,10,11_{In the cases reviewed by}

Enberg and Kaplan, 11 out of 46 patients (24%) had a history of blunt trauma to the back. It is postulated that presumably the formation of an epidural hema-toma then acts as a nidus for infection.1,10,11_However,

in the infant cases reviewed by Walter,1_{only 1 out of}

12 infants had a history of trauma to the spine.14

Adjacent vertebral osteomyelitis has been reported in 8% to 25% of children with SEA.1,12

Several authors implicate iatrogenic penetrating trauma such as a epidural anesthesia and LP as a risk factor for SEA.10,11,15_{However, a review of the}

litera-ture suggests that the risk of SEA associated with these procedures is extremely small. The largest re-view to date of infectious complications associated with epidural anesthesia in 1620 pediatric patients found only 1 SEA associated with a long-term tun-neled thoracic epidural catheter in an immunocom-promised patient with metastatic osteosarcoma.5

S aureus is the most frequently isolated etiologic agent in pediatric SEA, and was recovered in 54% of the cases reviewed by Enberg and Kaplan11_{and 75% of}

the infant cases reviewed by Walter et al.1_Other

organ-isms reported in pediatric SEA include Pneumococcus, otherStreptococcusspecies,Candidaand coliforms.

MRI is the diagnostic tool of choice for SEA. High resolution ultrasound is a useful, noninvasive alter-native that can be done at the bedside of the severely ill infant who may be too unstable to undergo imag-ing in an MRI scanner.3_{Laminectomy with surgical}

decompression and irrigation of the abscess cavity, plus systemic antibiotics for 4 to 8 weeks after drain-age is the traditional treatment for SEA. The duration of systemic antibiotics may need to be extended in cases of SEA with adjacent osteomyelitis.1_Some

au-thors support a limited laminectomy3,10,16 _to

mini-mize the subsequent risk of kyphosis. Walter et al1

reported the lone case where treatment via percuta-neous drainage was successful. Although this ap-proach offers the advantage of avoiding a laminec-tomy that can potentially lead to permanent spinal deformity in young infants and children, the authors concede that this should only be attempted if there is

no adjacent bony or disk involvement, or as an alter-native in a poor surgical candidate.

CONCLUSION

In summary, SEA in preverbal children is a rare but potentially devastating disease entity that often has a nonspecific presentation, requiring a high in-dex of suspicion for timely diagnosis and treatment. It should be considered in any young child with fever and spinal stiffness or tenderness, with or with-out neurologic deficits. It should also be considered when LP yields frank pus, when the cerebrospinal fluid (CSF) culture yieldsS aureus, or when the CSF findings are consistent with a parameningeal focus with sterile cultures. CSF findings that suggest a parameningeal focus include CSF pleocytosis, nega-tive Gram’s stain and culture, normal glucose, and elevated protein.17 _{Prompt surgical treatment and}

antibiotics should be undertaken to minimize the risk of progressive and permanent neurologic im-pairment. Our case illustrates that extensive spinal involvement may result in excellent outcomes and is the first to describe Currarino triad with SEA.

Kenneth A. Liu, MD* Jan D. Luhmann, MD*‡

*Department of Pediatrics ‡Division of Emergency Medicine

Washington University School of Medicine St Louis Children’s Hospital

St Louis, MO 63110-1077

REFERENCES

1. Walter RS, King JC Jr, Manley J, Rigamonti D. Spinal epidural abscess in infancy: successful percutaneous drainage in a nine-month-old and review of the literature.Pediatr Infect Dis J.1991;10:890 – 894

2. Marks WA, Bodensteiner JB. Anterior cervical epidural abscess with pneumococcus in an infant.J Child Neurol.1988;3:25–29

3. Gudinchet F, Chapuis L, Berger D. Diagnosis of anterior cervical spinal epidural abscess by US and MRI in a newborn.Pediatr Radiol.1991;21: 515–517

4. Firsching R, Frowein RA, Nittner K. Acute spinal epidural empyema: observations from seven cases.Acta Neurochir (Wien).

5. Strafford MA, Wilder RT, Berde CB. The risk of infection from epidural analgesia in children: a review of 1620 cases.Anesth Analg. 1995;80: 234 –238

6. Currarino G, Coln D, Votteler T. Triad of anorectal, sacral and presacral anomalies.AJR Am J Roentgenol.1981;137:395–398

7. Kochling J, Pistor G, Marzhauser Brands S, Nasir R, Lanksch WR. The Currarino syndrome— hereditary transmitted syndrome of anorectal, sacral and presacral anomalies. Case report and review of the literature. Eur J Pediatr Surg. 1996;6:114 –119

8. Lee SC, Chun YS, Jung SE, Park KW, Kim WK. Currarino triad: ano-rectal malformation, sacral bony abnormality, and presacral mass. A review of 11 cases. J Pediatr Surg.1997;32:58 – 61

9. Janneck C, Holthusen W. The Currarino triad—a study of 4 cases.Z Kinderchir. 1988;43:112–116

10. Fischer EG, Greene CS Jr, Winston KR. Spinal epidural abscess in children.Neurosurgery.1981;9:257–260

11. Enberg RN, Kaplan RJ. Spinal epidural abscess in children: early diag-nosis and immediate surgical drainage is essential to forestall paralysis. Clin Pediatr.1974;13:247–253

12. Rockney R, Ryan R, Knuckey N. Spinal epidural abscess: an infectious emergency. Clin Pediatr.1989;28:332–334

13. Batson OV. The function of the vertebral veins and their role in the spread of metastases.Clin Orthrop.1995;312:4 –9

14. Miller WH, Hesch JA. Nontuberculous spinal epidural abscess.Am J Dis Child.1962;104:269 –275

15. Bergman I, Wald ER, Meyer JD, Painter MJ. Epidural abscess and vertebral osteomyelitis following serial lumbar punctures.Pediatrics. 1983;72:476 – 480

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DOI: 10.1542/peds.104.5.1139

1999;104;1139

Pediatrics

Kenneth A. Liu and Jan D. Luhmann

Triad

Spinal Epidural Abscess in Preverbal Children: A Case Report With Currarino

Services

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