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Retropharyngeal abscess after BCG vaccination
International Journal of Pediatric Otorhinolaryngology Extra (2006) 1, 279—281
www.elsevier.com/locate/ijporl
CASE REPORT
Retropharyngeal abscess after BCG vaccination Nima Parvaneh a, Setareh Mamishi a,*, Maryam Monajemzadeh b a
Department of Pediatrics, Infectious Disease Research Center, Children’s Medical Center, Tehran University of Medical Sciences, Tehran, Iran b Department of Pathology, Children’s Medical Center, Tehran University of Medical Sciences, Tehran, Iran Received 19 June 2006; accepted 31 July 2006
KEYWORDS BCG vaccine; Complications; Retropharyngeal abscess
Summary Retropharyngeal abscess is a rare but serious disease in children. BCG infection is safe in immunocompetent hosts, but possible complications may occur. We report an 18-month-old girl who developed retropharyngeal abscess after BCG vaccination at birth. # 2006 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Retropharyngeal abscess (RPA), a rare disease in pediatric patients, is thought to be secondary to suppurative adenitis of the retropharyngeal lymph nodes. It is usually results from upper respiratory tract infection, contiguous infection or trauma [1]. It is a source of significant potential morbidity and mortality, if the diagnosis and management are delayed. Most retropharyngeal abscesses are polymicrobial in origin, with the dominant organisms being Streptococcus spp., Staphylococcus spp. and anaerobes [2]. Bacille Calmette-Gue ´rin (BCG) vaccine is a live vaccine made from attenuated strains of Mycobacterium bovis. In the vast majority of children, inoculation with BCG is harmless. However, a wide spectrum of adverse reactions has been observed [3]. Possible complications range from * Corresponding author at: No. 62 Gharib Street, Tehran, Iran. Tel.: +98 21 66 92 09 82; fax: +98 21 66 92 30 54. E-mail address: [email protected] (S. Mamishi).
local inflammatory reactions to disseminated BCG diseases and death [4]. We present the case of an 18-month-old girl with a retropharyngeal abscess caused by BCG and describe the clinical features of this very rare [5] but significant manifestation of BCG vaccination.
2. Case report An 18-month-old girl was seen in the emergency department because of the sub acute onset of progressive neck pain, swelling, dysphagia and drooling from one month prior to admission. The family denied previous upper respiratory tract infection, trauma or foreign body ingestion. Outpatient use of oral penicillin and cephalexin were ineffective. On the past history, she was the first child of a consanguineous marriage. She had received routine intradermal BCG vaccine just after birth. She had a history of draining pustular axillary and posterior auricular nodes at the age of 2 and 5 months, respectively.
1871-4048/$ — see front matter # 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.pedex.2006.07.008
280
N. Parvaneh et al.
Fig. 1 Computed tomography of the neck, showing bilateral retropharyngeal abscesses (white arrows).
On admission she was an acutely ill-looking girl with a pulse rate of 130 beats/min, respiratory rate of 40 breaths/min, and body temperature of 38.8 8C. Physical examination of the neck showed bilateral submandibular swelling and the girl was unable to move his neck because of severe pain. Oropharyngeal examination disclosed erythematous tonsils; no other abnormalities were noted. The physical examination findings revealed scars of previous draining abscesses on the right axillary and post auricular regions. A complete blood count revealed 15,600 white blood cells/ml with no band cells, neutrophils 60, lymphocytes 33 and monocytes 6% and hemoglobin 12.5 g/dl, and platelets 600 103/ml. Pediatric otolaryngologist to investigate the possibility of a neck abscess visited the patient. Due to the clinical suspicion of an RPA, a computed tomography (CT) scan was requested. The diagnosis of retropharyngeal abscess was established (Fig. 1) and the patient received open surgical incision and drainage through a transcervical approach due to impending airway obstruction. A
Fig. 2
pus specimen was obtained for Gram stain, aerobic and anaerobic cultures. Blood culture was also made. Smear and culture were negative for any bacterial agent. She was started on intravenous clindamycin and cefotaxime. On day 8 after admission, the patient’s fever continued, left neck pain worsened and neck swelling developed. CT of the neck showed low-density lesions in both right and left retropharyngeal spaces. Needle drainage under ultrasonographic guide yielded 20 ml of yellow pus. Bacterial, fungal and mycobacterial cultures and acid-fast staining were performed on the specimen. Numerous acid-fast bacilli were demonstrated on smears (Fig. 2), and cultures revealed growth of M. bovis, BCG strain. A test of delayed hypersensitivity to purified protein derivative (5 IU) was positive (a 15 mm induration). She was treated with isoniasid (15 mg/kg/day), rifampin (15 mg/kg/day), ethambutol (25 mg/kg/ day), and streptomycin (15 mg/kg/day). Interferon-g (50 mg/m2/week) was added as adjunctive. A search for underlying immunodeficiency revealed normal levels of serum immunoglobulins, normal NBT test, and normal number of peripheral blood lymphocyte subpopulations. HIV antibody test was negative by ELISA. The patient’s condition improved gradually, discharged on antimycobacterial therapy and interferon-g (IFN-g).
3. Discussion BCG, a live attenuated vaccine, has been used extensively for infants born in countries with a high incidence of Mycobacterium tuberculosis infection, and protects children from military tuberculosis and tuberculous meningitis [6].
Acid-fast bacilli demonstrated on Ziehl—Neelsen staining.
Retropharyngeal abscess after BCG vaccination It is considered a safe vaccine with a low incidence of complications. Adverse reactions associated with BCG vaccine have been well documented and are more common in younger vaccinees and with improper technique in vaccine administration. Adverse reactions also vary with the strain of BCG vaccination used and the method of administration [7]. The most frequent complications are local inflammatory reactions, including suppurative adenitis and localized abscess. Systemic adverse reactions to BCG vaccine are rare and include osteomyelitis and disseminated infection. Disseminated BCG infection has been estimated to develop in at a rate of one per million doses of administered vaccine and is associated with a high mortality [7]. Disseminated BCG usually occurs in children with underlying congenital or acquired immunodeficiency disorders such as severe combined immunodeficiency (SCID), chronic granulomatous disease (CGD), complete Di George syndrome, acquired immunodeficiency syndrome (AIDS) or Mendelian susceptibility to mycobacterial disease (MSMD), but only rarely in apparently normal individuals [8,9]. Disseminated BCG infection may involve the bones, joints, liver, spleen, and lymph nodes, and is usually fatal. We reported a patient who developed retropharyngeal abscess after BCG vaccination, a complication that is very rarely described [5]. The history of BCG adenitis, lack of associated infections with other organisms, positive test for delayed hypersensitivity to tuberculin and exclusion of classical immunodeficiencies made the diagnosis of MSMD very likely, however, we could not prove this at present. Molecular defects in IFN-g/interleukin-12 signaling are attributed to this syndrome [9]. Affected individuals develop severe clinical disease caused by weakly pathogenic mycobacterial species, such as BCG. The clinical phenotype ranges from fatal disseminated infection in early childhood to focal recurrent infection in adults [9]. Treatment of disseminated BCG infection is challenging, regarding underlying immunodeficiency and drug resistance of BCG strains [10]. Excluding the severe forms of immunodeficiencies (e.g. SCID or complete deficiency of IFNg receptor subunits) that are amenable only to stem cell transplantation, the other forms of
281 immunodeficiencies complicated with BCG disease should be treated with appropriate antimycobacterial regimens active against M. bovis and use of adjunctive IFN-g. Some authors advocate the use of life-long antimycobacterial prophylaxis as well [11]. Although retropharyngeal abscess caused by BCG is a very rare entity, high index of suspicion in the appropriate clinical setting and the rapid confirmation of M. bovis are essential to ensure correct management.
References [1] M. Coulthard, D. Isaacs, Retropharyngeal abscess, Arch. Dis. Child. 66 (1991) 1227—1230. [2] C.M. Philpott, D. Selvadurai, A.R. Banerjee, Paediatric retropharyngeal abscess, J. Laryngol. Otol. 118 (2004) 919—926. [3] J.L. Casanova, S. Blanche, J.F. Emile, et al., Idiopathic disseminated Bacillus Calmette-Guerin infection: a French national retrospective study, Pediatrics 98 (1996) 774—778. [4] J.M. Grange, Complications of Bacille Calmette-Guerin (BCG) vaccination and immunotherapy and their management, Commun. Dis. Public Health 1 (1998) 84—88. [5] S.A. Kostic-Joksic, Retropharyngeal abscess as unusual sequel of BCG vaccination, Tuberkuloza 5 (1953) 61—62. [6] G.A. Colditz, C.S. Berkey, F. Mosteller, et al., The efficacy of Bacillus Calmette-Guerin vaccination of newborns and infants in the prevention of tuberculosis: meta-analyses of the published literature, Pediatrics 96 (1995) 29—35. [7] A. Lotte, O. Wasz-Hockert, N. Poisson, et al., BCG complications. Estimates of the risks among vaccinated subjects and statistical analysis of their main characteristics, Adv. Tuberc. Res. 21 (1984) 107—193. [8] J.L. Casanova, E. Jouanguy, S. Lamhamedi, et al., Immunological conditions of children with BCG disseminated infection, Lancet 346 (1995) 581. [9] J.L. Casanova, L. Abel, Genetic dissection of immunity to mycobacteria: the human model, Annu. Rev. Immunol. 20 (2002) 581—620. [10] A.C. Hesseling, H.S. Schaaf, T. Victor, et al., Resistant Mycobacterium bovis Bacillus Calmette-Guerin disease: implications for management of Bacillus Calmette-Guerin Disease in human immunodeficiency virus-infected children, Pediatr. Infect. Dis. J. 23 (2004) 476—479. [11] M.H. Haverkamp, J.T. van Dissel, S.M. Holland, Human host genetic factors in non-tuberculous mycobacterial infection: lessons from single gene disorders affecting innate and adaptive immunity and lessons from molecular defects in interferon-gamma-dependent signaling, Microbes Infect. 8 (2006) 1157—1166.
www.elsevier.com/locate/ijporl
CASE REPORT
Retropharyngeal abscess after BCG vaccination Nima Parvaneh a, Setareh Mamishi a,*, Maryam Monajemzadeh b a
Department of Pediatrics, Infectious Disease Research Center, Children’s Medical Center, Tehran University of Medical Sciences, Tehran, Iran b Department of Pathology, Children’s Medical Center, Tehran University of Medical Sciences, Tehran, Iran Received 19 June 2006; accepted 31 July 2006
KEYWORDS BCG vaccine; Complications; Retropharyngeal abscess
Summary Retropharyngeal abscess is a rare but serious disease in children. BCG infection is safe in immunocompetent hosts, but possible complications may occur. We report an 18-month-old girl who developed retropharyngeal abscess after BCG vaccination at birth. # 2006 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Retropharyngeal abscess (RPA), a rare disease in pediatric patients, is thought to be secondary to suppurative adenitis of the retropharyngeal lymph nodes. It is usually results from upper respiratory tract infection, contiguous infection or trauma [1]. It is a source of significant potential morbidity and mortality, if the diagnosis and management are delayed. Most retropharyngeal abscesses are polymicrobial in origin, with the dominant organisms being Streptococcus spp., Staphylococcus spp. and anaerobes [2]. Bacille Calmette-Gue ´rin (BCG) vaccine is a live vaccine made from attenuated strains of Mycobacterium bovis. In the vast majority of children, inoculation with BCG is harmless. However, a wide spectrum of adverse reactions has been observed [3]. Possible complications range from * Corresponding author at: No. 62 Gharib Street, Tehran, Iran. Tel.: +98 21 66 92 09 82; fax: +98 21 66 92 30 54. E-mail address: [email protected] (S. Mamishi).
local inflammatory reactions to disseminated BCG diseases and death [4]. We present the case of an 18-month-old girl with a retropharyngeal abscess caused by BCG and describe the clinical features of this very rare [5] but significant manifestation of BCG vaccination.
2. Case report An 18-month-old girl was seen in the emergency department because of the sub acute onset of progressive neck pain, swelling, dysphagia and drooling from one month prior to admission. The family denied previous upper respiratory tract infection, trauma or foreign body ingestion. Outpatient use of oral penicillin and cephalexin were ineffective. On the past history, she was the first child of a consanguineous marriage. She had received routine intradermal BCG vaccine just after birth. She had a history of draining pustular axillary and posterior auricular nodes at the age of 2 and 5 months, respectively.
1871-4048/$ — see front matter # 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.pedex.2006.07.008
280
N. Parvaneh et al.
Fig. 1 Computed tomography of the neck, showing bilateral retropharyngeal abscesses (white arrows).
On admission she was an acutely ill-looking girl with a pulse rate of 130 beats/min, respiratory rate of 40 breaths/min, and body temperature of 38.8 8C. Physical examination of the neck showed bilateral submandibular swelling and the girl was unable to move his neck because of severe pain. Oropharyngeal examination disclosed erythematous tonsils; no other abnormalities were noted. The physical examination findings revealed scars of previous draining abscesses on the right axillary and post auricular regions. A complete blood count revealed 15,600 white blood cells/ml with no band cells, neutrophils 60, lymphocytes 33 and monocytes 6% and hemoglobin 12.5 g/dl, and platelets 600 103/ml. Pediatric otolaryngologist to investigate the possibility of a neck abscess visited the patient. Due to the clinical suspicion of an RPA, a computed tomography (CT) scan was requested. The diagnosis of retropharyngeal abscess was established (Fig. 1) and the patient received open surgical incision and drainage through a transcervical approach due to impending airway obstruction. A
Fig. 2
pus specimen was obtained for Gram stain, aerobic and anaerobic cultures. Blood culture was also made. Smear and culture were negative for any bacterial agent. She was started on intravenous clindamycin and cefotaxime. On day 8 after admission, the patient’s fever continued, left neck pain worsened and neck swelling developed. CT of the neck showed low-density lesions in both right and left retropharyngeal spaces. Needle drainage under ultrasonographic guide yielded 20 ml of yellow pus. Bacterial, fungal and mycobacterial cultures and acid-fast staining were performed on the specimen. Numerous acid-fast bacilli were demonstrated on smears (Fig. 2), and cultures revealed growth of M. bovis, BCG strain. A test of delayed hypersensitivity to purified protein derivative (5 IU) was positive (a 15 mm induration). She was treated with isoniasid (15 mg/kg/day), rifampin (15 mg/kg/day), ethambutol (25 mg/kg/ day), and streptomycin (15 mg/kg/day). Interferon-g (50 mg/m2/week) was added as adjunctive. A search for underlying immunodeficiency revealed normal levels of serum immunoglobulins, normal NBT test, and normal number of peripheral blood lymphocyte subpopulations. HIV antibody test was negative by ELISA. The patient’s condition improved gradually, discharged on antimycobacterial therapy and interferon-g (IFN-g).
3. Discussion BCG, a live attenuated vaccine, has been used extensively for infants born in countries with a high incidence of Mycobacterium tuberculosis infection, and protects children from military tuberculosis and tuberculous meningitis [6].
Acid-fast bacilli demonstrated on Ziehl—Neelsen staining.
Retropharyngeal abscess after BCG vaccination It is considered a safe vaccine with a low incidence of complications. Adverse reactions associated with BCG vaccine have been well documented and are more common in younger vaccinees and with improper technique in vaccine administration. Adverse reactions also vary with the strain of BCG vaccination used and the method of administration [7]. The most frequent complications are local inflammatory reactions, including suppurative adenitis and localized abscess. Systemic adverse reactions to BCG vaccine are rare and include osteomyelitis and disseminated infection. Disseminated BCG infection has been estimated to develop in at a rate of one per million doses of administered vaccine and is associated with a high mortality [7]. Disseminated BCG usually occurs in children with underlying congenital or acquired immunodeficiency disorders such as severe combined immunodeficiency (SCID), chronic granulomatous disease (CGD), complete Di George syndrome, acquired immunodeficiency syndrome (AIDS) or Mendelian susceptibility to mycobacterial disease (MSMD), but only rarely in apparently normal individuals [8,9]. Disseminated BCG infection may involve the bones, joints, liver, spleen, and lymph nodes, and is usually fatal. We reported a patient who developed retropharyngeal abscess after BCG vaccination, a complication that is very rarely described [5]. The history of BCG adenitis, lack of associated infections with other organisms, positive test for delayed hypersensitivity to tuberculin and exclusion of classical immunodeficiencies made the diagnosis of MSMD very likely, however, we could not prove this at present. Molecular defects in IFN-g/interleukin-12 signaling are attributed to this syndrome [9]. Affected individuals develop severe clinical disease caused by weakly pathogenic mycobacterial species, such as BCG. The clinical phenotype ranges from fatal disseminated infection in early childhood to focal recurrent infection in adults [9]. Treatment of disseminated BCG infection is challenging, regarding underlying immunodeficiency and drug resistance of BCG strains [10]. Excluding the severe forms of immunodeficiencies (e.g. SCID or complete deficiency of IFNg receptor subunits) that are amenable only to stem cell transplantation, the other forms of
281 immunodeficiencies complicated with BCG disease should be treated with appropriate antimycobacterial regimens active against M. bovis and use of adjunctive IFN-g. Some authors advocate the use of life-long antimycobacterial prophylaxis as well [11]. Although retropharyngeal abscess caused by BCG is a very rare entity, high index of suspicion in the appropriate clinical setting and the rapid confirmation of M. bovis are essential to ensure correct management.
References [1] M. Coulthard, D. Isaacs, Retropharyngeal abscess, Arch. Dis. Child. 66 (1991) 1227—1230. [2] C.M. Philpott, D. Selvadurai, A.R. Banerjee, Paediatric retropharyngeal abscess, J. Laryngol. Otol. 118 (2004) 919—926. [3] J.L. Casanova, S. Blanche, J.F. Emile, et al., Idiopathic disseminated Bacillus Calmette-Guerin infection: a French national retrospective study, Pediatrics 98 (1996) 774—778. [4] J.M. Grange, Complications of Bacille Calmette-Guerin (BCG) vaccination and immunotherapy and their management, Commun. Dis. Public Health 1 (1998) 84—88. [5] S.A. Kostic-Joksic, Retropharyngeal abscess as unusual sequel of BCG vaccination, Tuberkuloza 5 (1953) 61—62. [6] G.A. Colditz, C.S. Berkey, F. Mosteller, et al., The efficacy of Bacillus Calmette-Guerin vaccination of newborns and infants in the prevention of tuberculosis: meta-analyses of the published literature, Pediatrics 96 (1995) 29—35. [7] A. Lotte, O. Wasz-Hockert, N. Poisson, et al., BCG complications. Estimates of the risks among vaccinated subjects and statistical analysis of their main characteristics, Adv. Tuberc. Res. 21 (1984) 107—193. [8] J.L. Casanova, E. Jouanguy, S. Lamhamedi, et al., Immunological conditions of children with BCG disseminated infection, Lancet 346 (1995) 581. [9] J.L. Casanova, L. Abel, Genetic dissection of immunity to mycobacteria: the human model, Annu. Rev. Immunol. 20 (2002) 581—620. [10] A.C. Hesseling, H.S. Schaaf, T. Victor, et al., Resistant Mycobacterium bovis Bacillus Calmette-Guerin disease: implications for management of Bacillus Calmette-Guerin Disease in human immunodeficiency virus-infected children, Pediatr. Infect. Dis. J. 23 (2004) 476—479. [11] M.H. Haverkamp, J.T. van Dissel, S.M. Holland, Human host genetic factors in non-tuberculous mycobacterial infection: lessons from single gene disorders affecting innate and adaptive immunity and lessons from molecular defects in interferon-gamma-dependent signaling, Microbes Infect. 8 (2006) 1157—1166.