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Serratia marcescens Osteomyelitis in a  Newborn With Chronic  Granulomatous Disease

To the Editors:

Serratia marcescens infection

of bone and soft tissue is a presentation

of chronic granulomatous disease

(CGD) in infancy. 1 We report a case of a neonate diagnosed with X-CGD who developed a phalanx osteomyelitis due to S. marcenscens. A 25-day-old male was admitted

to our unit with a 2-day history of tender-

ness and swelling of the ring finger of the left hand. Topic therapy was administered without results. There was swelling between

the third and fourth fingers of the left hand

limiting movement of the metacarpophalan- geal joint. C reactive protein was 9.83 mg/ dL, and the leukocyte count was 20,810

cell/mm 3 with 13,260 cell/mm 3 neutrophils.

A radiograph showed osteomyelitis that

affected the proximal phalanx of the ring

finger; alternating areas of osteoscleroris and bone rarefaction were remarkable with

a initial exuberant process of periosteal

apposition (see Fig., Supplemental Digital Content 1, http://links.lww.com/INF/B536). The head of the phalanx was fractured. Cul- ture from the finger abscess grew S. marcen- scens. Intravenous antibiotic therapy with meropenem, amikacin and ciprofloxacin was given for 5 weeks. The family history was remarkable for the mother being a car-

rier of X-CGD. The brother was similarly affected. Molecular investigations led to the diagnosis of X-linked CGD in the infant. Marked clinical improvement was noted during the hospitalization, and he was dis- charged on prophylactic trimethoprim and itraconazole; oral ciprofloxacin was pre- scribed for an additional 2 months. Twelve months after the admission, the swelling had subsided and the bone had returned to

its normal contour, although bone expan-

sion was still detectable. The patient had

no functional sequelae. He is currently in

good clinical condition, receiving standard prophylaxis management and awaiting an

The authors have no funding or conflicts of interest to disclose. Supplemental digital content is available for this arti- cle. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website (www.pidj.com). Copyright © 2013 Lippincott Williams & Wilkins ISSN: 0891-3668/13/3208-0926 DOI: 10.1097/INF.0b013e31828f682a

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unrelated bone marrow transplantation from a suitably matched donor. Osteomyelitis occurs in 13% of CGD patients 2 ; in childhood, most occur in the long bones, with only 1–4% in the hands or feet. 3 Osteomyelitis in infancy particularly when caused by S. marcen- scens should raise the suspicion of the presence of CGD as an underlying condi- tion. We believe that this is the first case of osteomyelitis in a CGD patient reported in the neonatal period.

Irene Salfa, MD Nicoletta Cantarutti, MD Giulia Angelino, MD

Unit of Immunology and Infectious Disease University-Hospital Pediatric Department Bambino Gesù Children Hospital IRCCS

Gigliola Di Matteo, PhD Valentina Capo, BSc Giada Farinelli, PhD

Department of Public Health and Cellular Biology University of Rome “Tor Vergata”

Caterina Cancrini, MD, PhD

Unit of Immunology and Infectious Disease University-Hospital Pediatric Department Bambino Gesù Children Hospital IRCCS Rome, Italy

Alessandro Aiuti, MD, PhD

Unit of Immunology and Infectious Disease University Hospital Pediatric Department University of Rome “Tor Vergata” Children’s Hospital Bambino Gesù San Raffaele Telethon Institute for Gene Therapy San Raffaele Scientific Institute Milan, Italy

Paolo Palma, MD, PhD Andrea Finocchi, MD, PhD

Unit of Immunology and Infectious Disease University-Hospital Pediatric Department Bambino Gesù Children Hospital IRCCS Rome, Italy


1. Friend JC, Hilligoss DM, Marquesen M, et al. Skin ulcers and disseminated abscesses are characteristic of Serratia marcescens infection in older patients with chronic granulomatous disease. J Allergy Clin Immunol. 2009;124:


2. van den Berg JM, van Koppen E, Ahlin A, et al. Chronic granulomatous disease: the European experience. PLoS One. 2009;4:e5234.

3. Chad WM, Shahid B, Joseph AB, et al. Bahna Serratia marcescens osteomyelitis in an infant. Allergy Asthma Proc. 2006;27:544–548.

Management of Pediatric 

Contacts of Multidrug 

Resistant Tuberculosis in 

the United Kingdom

To the Editors:

C hildren exposed to drug-susceptible tuberculosis are less likely to progress

to active disease if receiving prophylactic

treatment, a practice now recommended by most international guidelines. 1,2 Appropriate prophylactic therapy cov- ering multidrug-resistant tuberculosis (MDR-TB) was shown to reduce the risk of progression to MDR-TB disease 5-fold in children who were household contacts of adults with pulmonary MDR-TB. 3 No unified guidance currently exists for management of MDR-TB–exposed children. 2,4 A retrospective audit of manage- ment of MDR-TB–exposed children was conducted as part of a wider survey of

management of pediatric MDR-TB in England. 5 Data were included if the con- tact had more than 8 hours of exposure to

a household or close social contact with

culture-confirmed pulmonary MDR-TB between January 1, 2006, and Decem- ber 31, 2010. Demographic, diagnostic and management details were collected through questionnaires sent to the lead TB pediatrician at each center. Returned

information was collated, and descriptive analyses were carried out using Excel ver- sion 97–2003 workbook. There were 23 children in 6 centers

in close contact with culture-proven pulmo-

nary MDR-TB. Eight were uninfected, did not receive chemoprophylaxis and to date none has developed disease. Twelve children were identified as latently infected. Eight children (66.6%) received treatment for presumed latent

MDR-TB. In all cases 2 drugs were used for

a median of 6 months (range 6–12 months).

Treatment regimens were selected accord- ing to the contacts’ in vitro susceptibility testing. Two children currently remain in follow-up for 2 years. The other 10 children were all well at the time of discharge with no evidence of TB disease. Three children had suspected but not culture-confirmed MDR-TB disease, all of whom had a known parental contact

NIHR Senior Fellowship to B.K. (MRC, Imperial College, UK). The authors have no other funding or conflicts of interest to disclose. Copyright © 2013 Lippincott Williams & Wilkins ISSN: 0891-3668/13/3208-0926 DOI: 10.1097/INF.0b013e31829157e9

The Pediatric Infectious Disease Journal  •  Volume 32, Number 8, August 2013

The Pediatric Infectious Disease Journal • Volume 32, Number 8, August 2013

Letters to the Editor

with MDR-TB. All children had sputum samples sent for cultures, but no organ- ism was isolated. The median number of drugs used for treatment was 4 (3–5), and the median planned length of therapy was 18 months (range 12–24). One of the 3 children received an injectable agent. The resistance pattern of the index case did not obviously account for the regimen choice in the children. All children were still being followed at the time of writ- ing, and none are known to be receiving directly observed treatment. Although we have a small dataset, our audit is the first to review the man- agement of pediatric contacts with infec- tious MDR-TB in England and is the largest dataset from Western Europe and North America published to date. Clini- cal practice showed considerable vari- ability, and children were less likely to receive prophylactic therapy if there was resistance to several drugs in the index case. Our small dataset cannot provide sufficient evidence for or against treat- ment for latent TB in MDR-exposed chil- dren, but illustrates that many clinicians chose to treat. It will be important to examine the age-related risk of progres- sion to active disease in larger groups of patients and balance these data with the likelihood of adherence, which might be compromised by treatment regimens sometimes more difficult to administer or prone to side-effects.

Bhanu Williams, MRCPCH

North West London Hospitals NHS Trust Harrow, United Kingdom

Shiva Ramroop, MRCPCH

Great Ormond Street Hospital

Pooja Shah, MB BS

Imperial NHS Trust

Laura Anderson, PhD

Health Protection Agency

Sreena Das, MRCPCH Anna Riddell, MRCPCH Susan Liebeschuetz, MRCPCH

Barts Health NHS Trust London, United Kingdom

Jolanta Bernatoniene, MRCPCH

Bristol Royal Hospital for Children Bristol, United Kingdom

Delane Shingadia, FRCPCH

Great Ormond Street Hospital

Beate Kampmann, PhD

Imperial College London, United Kingdom MRC Unit The Gambia

© 2013 Lippincott Williams & Wilkins


1. Beyers N, Gie RP, Schaaf HS, et al. A prospec- tive evaluation of children under the age of five years living in the same household as adults with recently diagnosed pulmonary tuberculo- sis. Int J Tuberc Lung Dis. 1997;1:38–43.

2. World Health Organization 2006. Guidelines for national tuberculosis programmes on the management of tuberculosis on children. Available at: http://www.who.int/tb/challenges/ children/en/. Accessed February 4, 2013.

3. Schaaf HS, Gie RP, Kennedy M, et al. Evaluation of young children in contact with adult multi- drug resistant pulmonary tuberculosis: a 30 month follow up. Pediatrics. 2002;109:765–771.

4. van der Werf MJ, Langendam MW, Sandgren A, et al. Lack of evidence to support policy development for management of contacts of MDR-TB patients: two systematic reviews. Int J Tuberc Lung Dis. 2012;16:288–296.

5. Williams B, Ramroop S, Shah P, et al. Multi-drug resistant tuberculosis in UK children: presenta- tion, management and outcome. Eur R J. 2013.

“Matching Michigan” in 

Neonatal Intensive Care 

Units: A Plea for Uniform 

Data Presentation

To the Editors:

W e thank Montgomery-Taylor and col- leagues 1 for reporting their experi-

ence of introducing “Matching Michigan”

(MM) to their neonatal intensive care unit.

MM was made a priority within the UK

National Health Service; reporting both infection and denominator data was recom- mended as the first step toward benchmark- ing and improving practices. 2 In a recent study 3 from 2 UK cent- ers over a 14-month period predating MM introduction, our definite catheter-related sepsis (CRS) rate was 6.8 per 1000 cath- eter days, somewhat higher than the 4.5 per 1000 catheter days reported by Montgom-

ery-Taylor et al using a similar case defini- tion. Our figures are not directly compara- ble because we only analyzed percutaneous central venous catheters (PCVCs). Inclu- sion of umbilical catheters probably lowers overall CRS rates by catheter days because umbilical lines are placed more frequently

than PCVCs, but are removed sooner and

therefore less liable to colonization/infec- tion. We suggest that future reporting of neonatal CRS rates should include subdi- visions by catheter type to facilitate valid comparisons between centers.

The authors have no funding or conflicts of interest to disclose. Copyright © 2013 by Lippincott Williams & Wilkins ISSN: 0891-3668/13/3208-0927 DOI: 10.1097/INF.0b013e318292f57f

The authors concluded that their

MM interventions had reduced all coag-

ulase-negative staphylococcal (CoNS) infections and blood culture (BC) contami- nations, and implied a reduction of cathe- ter-related bloodstream infection. 1 Due to a lack of comparable pre-MM data, it is not possible to say that this overall reduction represents a significant reduction in defi- nite CRS with MM. Fewer CoNS-positive BCs may simply reflect better hand hygiene

techniques reducing contaminant cultures rather than a true reduction in definite CRS. Some reduction in infection rates may also

have occurred coincidentally due to pre-

ceding and concurrent national improve-

ment efforts. 4

The authors did not include raw

data for total numbers of catheters inserted, catheter days, neonates admitted, or popu- lation demographics during their 18-month study period. Nor were data included for “catheter-suspected blood stream infec- tion” (catheter present but negative BC in the presence of antibiotics plus signs

of sepsis), even though such cases were defined for monthly collection via the National Patient Safety Agency MM form. 2 In our prospective study, 24 of 47 (51%) PCVCs removed from clinically septic

babies had an accompanying negative BC. 3 Provision of full denominator data is essen- tial for meaningful comparisons within and between neonatal centers.

The authors also excluded first day

CoNS-positive blood cultures, stating that “CoNS do not cause early onset sepsis.” 1 However, CoNS are a rare but recognized

cause of early-onset sepsis in neonates. 5

The authors found that non-CoNS

sepsis were “virtually all unrelated to the presence of a central catheter.” 1 Further analysis of our own dataset 3 shows that 2 of 15 (13%) definite CRS cases were non- CoNS: these comprised 2 (40%) of 5 non- CoNS septicemias present at line removal (n = 1 Enterococci, n = 1 Enterobacter clo- acae), and a non-CoNS organism was cul-

tured from almost 10% (6/61) of PCVCs

which had 1 culture-positive segment.

MM has shown a significant impact

in reducing infection rates in UK adult and

pediatric intensive care units. 4 As cath- eter care bundles are increasingly used in

NICUs, we consider it vital that all collect

a complete and consistent dataset, ideally

via established, dedicated neonatal data systems (such as the BadgerNet Platform [CleverMed Ltd, UK] and the Neonatal Data Analysis Unit [http://www.impe- rial.ac.uk/ndau], or the Vermont-Oxford Network’s program). Careful reporting of baseline denominator data will allow more meaningful epochal comparisons within

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