270 Case report
Salmonella osteomyelitis of the calcaneum bone in an immunocompetent child Mohamed Abdullaa, Yazan S. Khaleda,b,c and Harish Kapoora Salmonella osteomyelitis occurs infrequently in healthy children and can manifest in the subacute form. This condition has only been reported in few cases previously. We report the first case of primary subacute haematogenous osteomyelitis of the calcaneum in a healthy 12-year-old child. The patient made uneventful recovery following surgical drainage and antibiotic therapy. Histology of the tissue biopsy confirmed a diagnosis of Salmonella livingstone infection. Although the diagnosis of Salmonella osteomyelitis of the calcaneum can be difficult to establish, it should be considered as an aetiological factor even in healthy children. J Pediatr Orthop B
c 2014 Wolters Kluwer Health | Lippincott 23:270–273 Williams & Wilkins.
Introduction
difficulty in weight bearing of 3-weeks duration. He was initially presented to the accident and emergency (A&E) department following an injury to his left foot while playing football. Three weeks before this presentation, the patient had returned from Morocco where he developed an episode of self-limiting gastroenteritis. Radiographs of the left foot and ankle were taken and showed no visible fractures, and therefore the patient was treated conservatively and discharged. A blood test was not performed on this occasion as he was systemically well. Since then he has been experiencing difficulty in weight bearing. Two days before his hospitalization, he developed an increased level of pain and redness in his left heel with no associated history of pyrexia or rigors. Clinical examination in the A&E department showed an erythematous fluctuant blister, which was tender, at the base of the left heel. He had a normal range of active movement of the ankle with no evidence of neurovascular deficit. Laboratory investigations were normal including the inflammatory markers. He was admitted to a paediatric surgical ward with the diagnosis of cellulitis and was commenced on an intravenous antibiotic of flucloxacillin. The radiographs taken on this admission showed an epiphyseal reaction in the calcaneum. Subacute osteomyelitis of the calcaneum bone was diagnosed when a MRI of the left foot was performed for differential diagnosis as shown in Fig. 1. Although the MRI scan showed no abscess formation, the patient heel had partially discharged and an abscess had formed subsequently. Surgery was performed for the drainage of the abscess in the third day of admission, a day following the MRI report. During the operation, a 2 cm necrotic abscess cavity was found in communication with the calcaneum. The cavity was irrigated and debrided and curettage of the calcaneum was performed. Tissue biopsy and pus samples were taken for histology examination.
Salmonella osteomyelitis (SO) is a bacterial infection affecting the bone or bone marrow and can penetrate the bone through three main paths including bloodstream, from local areas of infection or through penetrating trauma including iatrogenic causes such as surgical fixation [1]. Staphylococcus aureus is the most common organism responsible for osteomyelitis in healthy children and adults [2]. Salmonella spp. are more common in immunocompromised children and in those who have sickle cell disease (SCD), but are rare in immunocompetent children [3]. The most common Salmonella spp. that are associated with osteomyelitis are Salmonella typhimurium, Salmonella paratyphi B and Salmonella enteritidis [4]. Primary subacute haematogenous osteomyelitis (PSHO) is a condition that is characterized by an insidious onset of pain in the affected limb, followed by a limp, reduced mobility and with mild or no systemic manifestations. The metaphyses of the long bones are the most commonly affected region by PSHO [5], whereas the epiphyseal region is rarely affected and is often associated with a delayed diagnosis [6]. In children, PSHO is considered as an important cause for a limp with an estimated rate of 2.4–27% of tarsal bones involvement [7]. Although the rate of osteomyelitis of the calcaneum was reported in 5–8% of all cases [6], there have been no reports on PSHO in the calcaneum secondary to Salmonella infection in children. In this report, we describe the surgical management of a 12-year-old child who was diagnosed with PSHO of the calcaneum bone, secondary to Salmonella spp., from a UK orthopaedic centre.
Patients and methods Patient details
A previously healthy 12-year-old child was referred to our trauma and orthopaedic department with a progressive c 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins 1060-152X
Journal of Pediatric Orthopaedics B 2014, 23:270–273 Keywords: calcaneum, primary subacute haematogenous osteomyelitis, Salmonella osteomyelitis a
Department of Trauma and Orthopaedic Surgery, Leeds General Infirmary, Section of Translational Anaesthetic and Surgical Sciences, University of Leeds and cSection of Translational Anaesthetic and Surgical Sciences, Leeds Institute of Molecular Medicine, Leeds, UK
b
Correspondence to Harish Kapoor, FRCS, MD, Department of Trauma and Orthopaedic Surgery, Leeds General Infirmary, Great George Street, Leeds LS1 3EX, UK Tel: + 44 113 243 2799; fax: + 44 113 243 7922; e-mail: [email protected]
DOI: 10.1097/BPB.0000000000000017
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Salmonella osteomyelitis of the calcaneum Abdulla et al.
271
Fig. 1
Fig. 2
MRI showing subacute osteomyelitis of the calcaneum bone.
MRI showing oedema within the posterior aspect of the calcaneum with no evidence of discrete collection.
Results The patient had an uneventful recovery postoperatively and was discharged home on postoperative day 8. The affected limb was immobilized in a below knee cast for 4 weeks and was continued on oral amoxicillin for the following 5 weeks. The histopathological examination of the tissue samples showed an infiltration with chronic inflammatory cells with no evidence of discrete granulomas or malignancy. The tissue and pus culture grew Salmonella livingstone. This strain is sensitive to amoxicillin/ ampicillin antibiotic and therefore the patient was commenced on intravenous amoxicillin of 1 g postoperatively for 1 week and was changed into oral amoxicillin for a total of 5 weeks of treatment. HIV serology was negative and haemoglobin electrophoresis was normal. Outcome and follow-up
The patient was weekly reviewed in the paediatric outpatient clinic by the operating consultant. His wound healed well and his inflammatory markers remained stable. He continued non-weight-bearing immobilization of the affected limb for 4 weeks. A follow-up radiograph at 2 weeks postoperatively showed bone sclerosis at the previous lytic areas of the calcaneum in keeping with healing. The patient was mobilized and fully weight bearing in the fifth week. At the 4-month follow-up, the patient complained of discomfort and a limp when walking over long distances. An MRI scan of the left ankle was performed, which demonstrated residual oedema within the middle and posterior aspect of the
calcaneum with no evidence of discrete collection as shown in Fig. 2. We decided to treat the symptoms conservatively and to continue with full weight bearing. At the 6-month follow-up, the patient remained well.
Discussion Salmonella osteomyelitis occurs very rarely in immunocompetent children. When established, however, it is often associated with pre-existing conditions such as SCD, connective tissue disorders, malignancy, surgical fixation and liver disease [8–10]. Although S. aureus remains a more common cause of osteomyelitis in this type of patients, their immunodeficiency can leave them susceptible to SO [11]. There are different types of Salmonella spp. that can cause osteomyelitis; the strains most commonly encountered are S. typhimurium, S. paratyphi B and S. enteritidis [4]. Type B is estimated to be the most common cause in children who have no haematological abnormalities [12], whereas nontyphoid Salmonella bacteraemia occurs after an episode of gastroenteritis [13]. Our patient was of British descent and the organism type identified was S. livingstone. This organism is mainly found in food products, including poultry and eggs [14] and the organism was first isolated in a patient with gastroenteritis staying at a hotel in Victoria Falls in 1951 [15]. Outbreaks of S. livingstone infection have been rarely reported in Europe, with an increased number of isolated S. livingstone species in Tunisia and North Africa [16]. Salmonella livingstone bacteraemia is predisposed by old age, pre-existing gastrointestinal disorders or other alterations of
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
272 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 3
the gastrointestinal environment that could reduce resistance to Salmonella infection [17]. Our patient had a recent travelling history to North Africa where he developed signs and symptoms of gastroenteritis. Although the patient was treated conservatively for the gastroenteritis in Morocco, stool cultures were not taken to isolate the causing species and therefore no antibiotics were given. This is the likely explanation for the PSHO of the calcaneum in this healthy child. Our patient had a 3-week history of discomfort at the heel, developed a mild limp but remained systemically stable in keeping with PSHO. Misdiagnosis of PSHO in immunocompetent children can occur frequently because of the nonspecific symptoms at presentation and the absence of predisposing conditions such as SCD [1,18]. There are often no specific blood tests to confirm the diagnosis, except clinical suspicion and positive MRI scan [19]. Diagnosis is often confirmed on histology, showing cells of inflammation and granulation [4]. In our case, the organism was identified only on tissue sample cultures taken during curettage. It has been reported, when cultures are taken from curettage, that S. livingstone organisms are found to be positive in 31–83% of all cases in all types of osteomyelitis [20]. Others reported that when antibiotic treatment is started it is less likely to find positive blood cultures, which was evident in our case [21]. A delay in confirming the diagnosis and instigating the appropriate management can lead to an irreversible damage of the growth plates [22]. Our patient was discharged home following his first presentation to the A&E department due to the lack of specific symptoms and low suspicion for PSHO. However, during his hospitalization he was diagnosed in a timely Table 1
manner and was commenced on intravenous antibiotic, which was adjusted to culture findings. The incidence of osteomyelitis affecting the tarsal bones in immunocompetent children has been reported to be 2.4–27% [7,23], whereas the rate of calcaneum osteomyelitis is between 5 and 8% of all reported cases [1,24,25]. The involvement of these small bones often manifest in the subacute form [26]. PSHO with Salmonella infection has been reported in other rare sites, such as the pelvis, the tibia, the navicular and the distal femur in immunocompetent children [3,4,27,28]. The characteristic features of the disease and the treatments for the four case reports are outlined in Tables 1 and 2. Although some authors reported treating PSHO with antibiotic alone [7,29,30], we opted to perform surgical drainage and tissue debridement due to the formation of an abscess, weak response to antibiotics and lack of confirmed diagnosis. Our practice is to start antibiotics after surgery or tissue biopsy unless the patient is presented with systemic manifestations. Our patient received a total of 7 weeks of antibiotics, 2 weeks intravenously followed by 5 weeks of oral antibiotic. While there remains lack of sufficient evidence to support the timing of surgery for PSHO, the length of antibiotics use is also controversial. Nonetheless, surgery is indicated in any patient who develops signs of abscess formation or has not responded to antibiotic treatment [31].
Conclusion
Salmonella osteomyelitis occurs rarely in immunocompetent children. Similarly, PSHO occurs infrequently in the
Patients demographic details and disease characteristics
References Canessa et al. [3] Lebeaux et al. [27] Celik et al. [4] Abdelgawad et al. [28]
Age (years)
Sex
Presentation
11 11
Male Female
Fever, headache, lumbar pain and limp 4 weeks leg pain, erythema, oedema and fluctuating abscess Limping and pain, gastroenteritis 10 days previously Fever, unable to bear weight, knee effusion
4
Male
1.5
Male
Site Pelvis Tibia
Diagnostic tools
Navicular
MRI and CT Ultrasound and MRI MRI
Distal femur
MRI
CT, computed tomography; MRI, magnetic resonance imaging.
Table 2
Management methods and follow-up
References
Organism
Treatment option
Antibiotic
Canessa et al. [3]
Salmonella typhimurium
Antibiotics for 6 months
Levofloxacin and linezolid
Celik et al. [4]
Salmonella enteritidis
Cefotaxime
Lebeaux et al. [27]
Salmonella colindale
Surgical debridement and antibiotics for 5 weeks Surgical washout and 6 weeks antibiotics
Abdelgawad et al. [28]
Salmonella type B
CT guided drainage and 6 weeks antibiotics
Intravenous ceftriaxone for 7 days and oral ofloxacin for 6 weeks Intravenous ceftriaxone
Follow-up MRI at 6 months showed improvement Good resolution 2-year follow-up good resolution 2-month follow-up showed good healing
CT, computed tomography; MRI, magnetic resonance imaging.
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Salmonella osteomyelitis of the calcaneum Abdulla et al. 273
epiphysis and the tarsal bones. Nonetheless, clinicians should consider SO of the calcaneum even in healthy children who may present with normal inflammatory markers and normal radiographic images and with a recent history of gastroenteritis.
15 16
17
Acknowledgements Conflicts of interest
18
There are no conflicts of interest.
19
References
20
1
2 3
4
5 6
7 8 9
10 11 12
13 14
Waldvogel FA, Medoff G, Swarts MN. Osteomyelitis: review of the clinical features, therapeutic considerations and unusual aspects. N Engl J Med 1970; 282:198–206. Kaplan SL. Osteomyelitis in children. Infect Dis Clin North Am 2005; 19:787–797. Canessa C, Trapani S, Campanacci D, Chiappini E, Maglione M, Resti M. Salmonella pelvic osteomyelitis in an immunocompetent child. BMJ Case Rep 2011; 2011:pii: bcr0220113831. Celik FC, Sayli TR, Ocguder DA, Bozkurt M, Okdemir D. Primary subacute Salmonella osteomyelitis of the navicular bone in a child with normal immunity. J Pediatr Orthop B 2009; 18:225–227. Lazzarini L, Mader JT, Calhoun JH. Osteomyelitis in long bones. J Bone Joint Surg Am 2004; 86:2305–2318. Kelsey R, Kor A, Cordano F. Haematogenous osteomyelitis of the calcaneus in children: surgical treatment and use of implanted antibiotic beads. J Foot Ankle Surg 1995; 34:547–555. Ezra E, Wientroub S. Primary subacute haematogenous osteomyelitis of the tarsal bones in children. J Bone Joint Surg Br 1997; 79:983–986. Cherubin CE, Neu HC, Imperato PJ, Harvey RP, Bellen N. Septicemia with non-typhoid Salmonella. Medicine (Baltimore) 1974; 53:365–376. Adeyokunnu AA, Hendrickse RG. Salmonella osteomyelitis in childhood. A report of 63 cases seen in Nigerian children of whom 57 had sickle cell anaemia. Arch Dis Child 1980; 55:175–184. Higgins GA, Sandiford JA, Blair WC. Remote Salmonella infections. World J Surg 1982; 6:236–240. Klein JD, Leach KA. Pediatric pelvic osteomyelitis. Clin Pediatr (Phila) 2007; 46:787–790. Sucato DJ, Gillespie R. Salmonella pelvic osteomyelitis in normal children: report of two cases and a review of the literature. J Pediatr Orthop 1997; 17:463–466. Adlam C, Pearce JH, Smith H. Virulence mechanisms of staphylococci grown in vivo and in vitro. J Med Microbiol 1970; 3:147–155. Odongo MO, McLaren IM, Smith JE, Wray C. A biotyping scheme for Salmonella livingstone. Br Vet J 1990; 146:75–79.
21
22
23 24 25 26 27
28
29 30
31
Picton WH, Stirrup W. A new Salmonella type, Salm. livingstone. J Pathol Bacteriol 1953; 66:310–312. Guerin PJ, De Jong B, Heir E, Hasseltvedt V, Kapperud G, Styrmo K, et al. Outbreak of Salmonella livingstone infection in Norway and Sweden due to contaminated processed fish products. Epidemiol Infect 2004; 132:889–895. Old DC, Porter-Boveri M, Munro DS. Human infection in Tayside, Scotland due to Salmonella serotype livingstone. J Med Microbiol 1994; 40:134–140. Fox IM, Aponte JM. Hematogenous osteomyelitis of the calcaneus. J Am Podiatr Med Assoc 1993; 83:681–684. Roberts JM, Drummond DS, Breed AL, Chesney J. Subacute hematogeneous osteomyelitis in children: a retrospective study. J Pediatr Orthop 1982; 2:249–254. Verdier I, Gayet-Ageron A, Ploton C, Taylor P, Benito Y, Freydiere AM, et al. Contribution of a broad range polymerase chain reaction to the diagnosis of osteoarticular infections caused by Kingella kingae: description of twenty-four recent pediatric diagnoses. Pediatr Infect Dis J 2005; 24:692–696. Christiansen P, Frederiksen B, Glazowski J, Scavenius M, Knudsen FU. Epidemiologic, bacteriologic, and long-term follow-up data of children with acute hematogenous osteomyelitis and septic arthritis: a ten-year review. J Pediatr Orthop B 1999; 8:302–305. Borris LC, Helleland H. Growth disturbance of the hind part of the foot following osteomyelitis of the calcaneus in the newborn. J Bone Joint Surg 1985; 68:302–305. White M, Denison WM. Acute haematogenous osteomyelitis in childhood: a review of 212 cases. J Bone Joint Surg 1952; 34:608–623. Wang EM, Simpson S, Bennet GC. Osteomyelitis of the calcaneum. J Bone Joint Surg 1992; 74:906–909. Feigin RD, McAlister WH, Joaquin VH, Middelkamp JN. Osteomyelitis of the calcaneus. Report of eight cases. Am J Dis Child 1970; 119:61–65. Trueta J, Morgan JD. Late results in the treatment of one hundred cases of acute haematogenous osteomyelitis. Br J Surg 1954; 41:449–457. Lebeaux D, Zarrouk V, Petrover D, Nicolas-Chanoine MH, Fantin B. Salmonella colindale osteomyelitis in an immunocompetent female patient. Med Mal Infect 2012; 42:36–37. Abdelgawad AA, Rybak LD, Sheth M, Rabinowitz SS, Jayaram N, Sala DA, van Bosse HJ. Treatment of acute Salmonella epiphyseal osteomyelitis using computed tomography-guided drainage in a child without sickle cell disease. J Pediatr Orthop B 2007; 16:415–418. Ross ER, Cole WG. Treatment of subacute osteomyelitis in childhood. J Bone Joint Surg Br 1985; 67:443–448. Hamdy RC, Lawton L, Carey T, Wiley J, Marton D. Subacute hematogenous osteomyelitis: are biopsy and surgery always indicated? J Pediatr Orthop 1996; 16:220–223. Mustafa MM, Saez-Llorens X, McCracken GH Jr, Nelson JD. Acute hematogenous pelvic osteomyelitis in infants and children. Pediatr Infect Dis J 1990; 9:416–421.
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Salmonella osteomyelitis of the calcaneum bone in an immunocompetent child Mohamed Abdullaa, Yazan S. Khaleda,b,c and Harish Kapoora Salmonella osteomyelitis occurs infrequently in healthy children and can manifest in the subacute form. This condition has only been reported in few cases previously. We report the first case of primary subacute haematogenous osteomyelitis of the calcaneum in a healthy 12-year-old child. The patient made uneventful recovery following surgical drainage and antibiotic therapy. Histology of the tissue biopsy confirmed a diagnosis of Salmonella livingstone infection. Although the diagnosis of Salmonella osteomyelitis of the calcaneum can be difficult to establish, it should be considered as an aetiological factor even in healthy children. J Pediatr Orthop B
c 2014 Wolters Kluwer Health | Lippincott 23:270–273 Williams & Wilkins.
Introduction
difficulty in weight bearing of 3-weeks duration. He was initially presented to the accident and emergency (A&E) department following an injury to his left foot while playing football. Three weeks before this presentation, the patient had returned from Morocco where he developed an episode of self-limiting gastroenteritis. Radiographs of the left foot and ankle were taken and showed no visible fractures, and therefore the patient was treated conservatively and discharged. A blood test was not performed on this occasion as he was systemically well. Since then he has been experiencing difficulty in weight bearing. Two days before his hospitalization, he developed an increased level of pain and redness in his left heel with no associated history of pyrexia or rigors. Clinical examination in the A&E department showed an erythematous fluctuant blister, which was tender, at the base of the left heel. He had a normal range of active movement of the ankle with no evidence of neurovascular deficit. Laboratory investigations were normal including the inflammatory markers. He was admitted to a paediatric surgical ward with the diagnosis of cellulitis and was commenced on an intravenous antibiotic of flucloxacillin. The radiographs taken on this admission showed an epiphyseal reaction in the calcaneum. Subacute osteomyelitis of the calcaneum bone was diagnosed when a MRI of the left foot was performed for differential diagnosis as shown in Fig. 1. Although the MRI scan showed no abscess formation, the patient heel had partially discharged and an abscess had formed subsequently. Surgery was performed for the drainage of the abscess in the third day of admission, a day following the MRI report. During the operation, a 2 cm necrotic abscess cavity was found in communication with the calcaneum. The cavity was irrigated and debrided and curettage of the calcaneum was performed. Tissue biopsy and pus samples were taken for histology examination.
Salmonella osteomyelitis (SO) is a bacterial infection affecting the bone or bone marrow and can penetrate the bone through three main paths including bloodstream, from local areas of infection or through penetrating trauma including iatrogenic causes such as surgical fixation [1]. Staphylococcus aureus is the most common organism responsible for osteomyelitis in healthy children and adults [2]. Salmonella spp. are more common in immunocompromised children and in those who have sickle cell disease (SCD), but are rare in immunocompetent children [3]. The most common Salmonella spp. that are associated with osteomyelitis are Salmonella typhimurium, Salmonella paratyphi B and Salmonella enteritidis [4]. Primary subacute haematogenous osteomyelitis (PSHO) is a condition that is characterized by an insidious onset of pain in the affected limb, followed by a limp, reduced mobility and with mild or no systemic manifestations. The metaphyses of the long bones are the most commonly affected region by PSHO [5], whereas the epiphyseal region is rarely affected and is often associated with a delayed diagnosis [6]. In children, PSHO is considered as an important cause for a limp with an estimated rate of 2.4–27% of tarsal bones involvement [7]. Although the rate of osteomyelitis of the calcaneum was reported in 5–8% of all cases [6], there have been no reports on PSHO in the calcaneum secondary to Salmonella infection in children. In this report, we describe the surgical management of a 12-year-old child who was diagnosed with PSHO of the calcaneum bone, secondary to Salmonella spp., from a UK orthopaedic centre.
Patients and methods Patient details
A previously healthy 12-year-old child was referred to our trauma and orthopaedic department with a progressive c 2014 Wolters Kluwer Health | Lippincott Williams & Wilkins 1060-152X
Journal of Pediatric Orthopaedics B 2014, 23:270–273 Keywords: calcaneum, primary subacute haematogenous osteomyelitis, Salmonella osteomyelitis a
Department of Trauma and Orthopaedic Surgery, Leeds General Infirmary, Section of Translational Anaesthetic and Surgical Sciences, University of Leeds and cSection of Translational Anaesthetic and Surgical Sciences, Leeds Institute of Molecular Medicine, Leeds, UK
b
Correspondence to Harish Kapoor, FRCS, MD, Department of Trauma and Orthopaedic Surgery, Leeds General Infirmary, Great George Street, Leeds LS1 3EX, UK Tel: + 44 113 243 2799; fax: + 44 113 243 7922; e-mail: [email protected]
DOI: 10.1097/BPB.0000000000000017
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Salmonella osteomyelitis of the calcaneum Abdulla et al.
271
Fig. 1
Fig. 2
MRI showing subacute osteomyelitis of the calcaneum bone.
MRI showing oedema within the posterior aspect of the calcaneum with no evidence of discrete collection.
Results The patient had an uneventful recovery postoperatively and was discharged home on postoperative day 8. The affected limb was immobilized in a below knee cast for 4 weeks and was continued on oral amoxicillin for the following 5 weeks. The histopathological examination of the tissue samples showed an infiltration with chronic inflammatory cells with no evidence of discrete granulomas or malignancy. The tissue and pus culture grew Salmonella livingstone. This strain is sensitive to amoxicillin/ ampicillin antibiotic and therefore the patient was commenced on intravenous amoxicillin of 1 g postoperatively for 1 week and was changed into oral amoxicillin for a total of 5 weeks of treatment. HIV serology was negative and haemoglobin electrophoresis was normal. Outcome and follow-up
The patient was weekly reviewed in the paediatric outpatient clinic by the operating consultant. His wound healed well and his inflammatory markers remained stable. He continued non-weight-bearing immobilization of the affected limb for 4 weeks. A follow-up radiograph at 2 weeks postoperatively showed bone sclerosis at the previous lytic areas of the calcaneum in keeping with healing. The patient was mobilized and fully weight bearing in the fifth week. At the 4-month follow-up, the patient complained of discomfort and a limp when walking over long distances. An MRI scan of the left ankle was performed, which demonstrated residual oedema within the middle and posterior aspect of the
calcaneum with no evidence of discrete collection as shown in Fig. 2. We decided to treat the symptoms conservatively and to continue with full weight bearing. At the 6-month follow-up, the patient remained well.
Discussion Salmonella osteomyelitis occurs very rarely in immunocompetent children. When established, however, it is often associated with pre-existing conditions such as SCD, connective tissue disorders, malignancy, surgical fixation and liver disease [8–10]. Although S. aureus remains a more common cause of osteomyelitis in this type of patients, their immunodeficiency can leave them susceptible to SO [11]. There are different types of Salmonella spp. that can cause osteomyelitis; the strains most commonly encountered are S. typhimurium, S. paratyphi B and S. enteritidis [4]. Type B is estimated to be the most common cause in children who have no haematological abnormalities [12], whereas nontyphoid Salmonella bacteraemia occurs after an episode of gastroenteritis [13]. Our patient was of British descent and the organism type identified was S. livingstone. This organism is mainly found in food products, including poultry and eggs [14] and the organism was first isolated in a patient with gastroenteritis staying at a hotel in Victoria Falls in 1951 [15]. Outbreaks of S. livingstone infection have been rarely reported in Europe, with an increased number of isolated S. livingstone species in Tunisia and North Africa [16]. Salmonella livingstone bacteraemia is predisposed by old age, pre-existing gastrointestinal disorders or other alterations of
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
272 Journal of Pediatric Orthopaedics B 2014, Vol 23 No 3
the gastrointestinal environment that could reduce resistance to Salmonella infection [17]. Our patient had a recent travelling history to North Africa where he developed signs and symptoms of gastroenteritis. Although the patient was treated conservatively for the gastroenteritis in Morocco, stool cultures were not taken to isolate the causing species and therefore no antibiotics were given. This is the likely explanation for the PSHO of the calcaneum in this healthy child. Our patient had a 3-week history of discomfort at the heel, developed a mild limp but remained systemically stable in keeping with PSHO. Misdiagnosis of PSHO in immunocompetent children can occur frequently because of the nonspecific symptoms at presentation and the absence of predisposing conditions such as SCD [1,18]. There are often no specific blood tests to confirm the diagnosis, except clinical suspicion and positive MRI scan [19]. Diagnosis is often confirmed on histology, showing cells of inflammation and granulation [4]. In our case, the organism was identified only on tissue sample cultures taken during curettage. It has been reported, when cultures are taken from curettage, that S. livingstone organisms are found to be positive in 31–83% of all cases in all types of osteomyelitis [20]. Others reported that when antibiotic treatment is started it is less likely to find positive blood cultures, which was evident in our case [21]. A delay in confirming the diagnosis and instigating the appropriate management can lead to an irreversible damage of the growth plates [22]. Our patient was discharged home following his first presentation to the A&E department due to the lack of specific symptoms and low suspicion for PSHO. However, during his hospitalization he was diagnosed in a timely Table 1
manner and was commenced on intravenous antibiotic, which was adjusted to culture findings. The incidence of osteomyelitis affecting the tarsal bones in immunocompetent children has been reported to be 2.4–27% [7,23], whereas the rate of calcaneum osteomyelitis is between 5 and 8% of all reported cases [1,24,25]. The involvement of these small bones often manifest in the subacute form [26]. PSHO with Salmonella infection has been reported in other rare sites, such as the pelvis, the tibia, the navicular and the distal femur in immunocompetent children [3,4,27,28]. The characteristic features of the disease and the treatments for the four case reports are outlined in Tables 1 and 2. Although some authors reported treating PSHO with antibiotic alone [7,29,30], we opted to perform surgical drainage and tissue debridement due to the formation of an abscess, weak response to antibiotics and lack of confirmed diagnosis. Our practice is to start antibiotics after surgery or tissue biopsy unless the patient is presented with systemic manifestations. Our patient received a total of 7 weeks of antibiotics, 2 weeks intravenously followed by 5 weeks of oral antibiotic. While there remains lack of sufficient evidence to support the timing of surgery for PSHO, the length of antibiotics use is also controversial. Nonetheless, surgery is indicated in any patient who develops signs of abscess formation or has not responded to antibiotic treatment [31].
Conclusion
Salmonella osteomyelitis occurs rarely in immunocompetent children. Similarly, PSHO occurs infrequently in the
Patients demographic details and disease characteristics
References Canessa et al. [3] Lebeaux et al. [27] Celik et al. [4] Abdelgawad et al. [28]
Age (years)
Sex
Presentation
11 11
Male Female
Fever, headache, lumbar pain and limp 4 weeks leg pain, erythema, oedema and fluctuating abscess Limping and pain, gastroenteritis 10 days previously Fever, unable to bear weight, knee effusion
4
Male
1.5
Male
Site Pelvis Tibia
Diagnostic tools
Navicular
MRI and CT Ultrasound and MRI MRI
Distal femur
MRI
CT, computed tomography; MRI, magnetic resonance imaging.
Table 2
Management methods and follow-up
References
Organism
Treatment option
Antibiotic
Canessa et al. [3]
Salmonella typhimurium
Antibiotics for 6 months
Levofloxacin and linezolid
Celik et al. [4]
Salmonella enteritidis
Cefotaxime
Lebeaux et al. [27]
Salmonella colindale
Surgical debridement and antibiotics for 5 weeks Surgical washout and 6 weeks antibiotics
Abdelgawad et al. [28]
Salmonella type B
CT guided drainage and 6 weeks antibiotics
Intravenous ceftriaxone for 7 days and oral ofloxacin for 6 weeks Intravenous ceftriaxone
Follow-up MRI at 6 months showed improvement Good resolution 2-year follow-up good resolution 2-month follow-up showed good healing
CT, computed tomography; MRI, magnetic resonance imaging.
Copyright © Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Salmonella osteomyelitis of the calcaneum Abdulla et al. 273
epiphysis and the tarsal bones. Nonetheless, clinicians should consider SO of the calcaneum even in healthy children who may present with normal inflammatory markers and normal radiographic images and with a recent history of gastroenteritis.
15 16
17
Acknowledgements Conflicts of interest
18
There are no conflicts of interest.
19
References
20
1
2 3
4
5 6
7 8 9
10 11 12
13 14
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