• View in gallery

    Case 1: An ostempyesis that introduces into the thorax with compression of the pleura without lung involvement or pleural effusion can be observed (A). In the reconstruction, the destruction of the final portion of the fifth costal arc can be observed (B).

  • View in gallery

    Case 2: Reconstruction in 3D with partial destruction of the left 10th costal arc in the final portion (A). Computed tomography scan with the presence of mass dependent on the left costal grid, presence of lytic lesions, and liquid collection with infiltration of lung and pleural tissue (B).

  • View in gallery

    Case 3: Chest X-ray with a lytic lesion simulating an expansive bony lesion (A) on the fifth rib measuring 200 mm2. Computed tomography scan with a bone lytic lesion on the anterolateral side of the same rib of 3 cm in length (B) and 1 cm in width (C).

  • 1.

    Rafiqi K, Yousri B, Arihi M, Bjitro C, Aboumaarouf M, El Andaloussi M, 2013. Unusual locations of osteoarticular tuberculosis in children: a report of 12 cases. Orthop Traumatol Surg Res 99: 347351.

    • Search Google Scholar
    • Export Citation
  • 2.

    National Epidemiological Surveillance System (SINAVE) of Mexico, 2015. Epidemiological Profile of Tuberculosis in Mexico [Perfil Epidemiológico de la Tuberculosis en México]. Health Ministry, SINAVE.

    • Search Google Scholar
    • Export Citation
  • 3.

    Agrawal V, Joshi MK, Jain BK, Mohanty D, Gupta A, 2008. Tuberculotic osteomyelitis of rib-a surgical entity. Interact Cardiovasc Thorac Surg 7: 10281030.

    • Search Google Scholar
    • Export Citation
  • 4.

    Kritsaneepaiboon S, Andres MM, Tatco VR, Lim CC, Concepción ND, 2017. Extrapulmonary involvement in pediatric tuberculosis. Pediatr Radiol 47: 12491259.

    • Search Google Scholar
    • Export Citation
  • 5.

    Grover SB, Jain M, Dumeer S, Sirari N, Bansal M, Badgujar D, 2011. Chest wall tuberculosis—a clinical and imaging experience. Indian J Radiol Imaging 21: 2833.

    • Search Google Scholar
    • Export Citation
  • 6.

    Chiu NC, 2015. Mycobacterium bovis bcg-associated osteomielitis/osteítis, Taiwan. Emerg Infect Dis 21: 539540.

  • 7.

    Terreri MT, Yamada AF, 2008. Osteitis caused by BCG vaccination. Pediatr Radiol 38: 481.

  • 8.

    Lee SH, Abramson SB, 1996. Infections of the musculoskeletal system by M. tuberculosis. Rom WN, Garay SM, eds. Tuberculosis. New York, NY: Little Brown and Company, 635644.

    • Search Google Scholar
    • Export Citation
  • 9.

    Faure E, Souilamas R, Riquet M, 1998. Cold abscess of the chest wall: a surgical entity? Ann Thorac Surg 66: 11741178.

  • 10.

    Chang DS, Rafii M, McGuinness G, 1999. Diagnostic issues in tuberculosis of the ribs with a review of 12 surgically proven cases. Respirology 4: 249253.

    • Search Google Scholar
    • Export Citation
  • 11.

    Perez-Velez C, 2012. Pediatric tuberculosis: new guidelines and recommendations. Curr Opin Pediatr 24: 319328.

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Case Report: Chest Wall Tuberculosis without Pulmonary Involvement in Three Pediatric Immunocompetent Patients

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  • 1 Service of Infectology, National Institute of Pediatrics (NIP), Mexico City, Mexico;
  • | 2 Medical Direction, National Institute of Pediatrics, Mexico City, Mexico;
  • | 3 Service of Radiology, National Institute of Pediatrics, Mexico City, Mexico;
  • | 4 Pediatric Infectious Diseases, Cafam Clinic, Bogota, Colombia;
  • | 5 Laboratory of Pharmacology, National Institute of Pediatrics, Mexico City, Mexico;
  • | 6 Department of Pharmacology, Faculty of Medicine, National Autonomous University of Mexico, Mexico City, Mexico;
  • | 7 Service of Traumatology and Orthopedics, National Institute of Pediatrics, Mexico City, Mexico

Primary rib cage tuberculosis (TB) is an infrequent form of presentation and represents 1% of all cases of osteoarticular TB. We report three cases of children who were previously healthy and who began with swelling of the anterior surface of the rib as initial manifestation of TB. The most important clinical presentations in this series were swelling and pain, with lytic lesions and a soft tissue mass in image studies simulating oncologic pathologies. Because none of the cases had positive epidemiological contact, TB was initially not considered, so the delay in diagnosis from the onset of symptoms was 4, 1, and 2 months, respectively. The diagnosis was made through histomorphological analyses. Treatment was administered during 12, 10, and 9 months. Posttreatment studies did not show any evidence of extrapulmonary TB and until date, the patients remained without relapse or active disease. The findings in our cases illustrate that the diagnosis of chest wall TB should be suspected in all patients from endemic areas who present rib injury.

INTRODUCTION

Tuberculosis (TB) continues to be an important world health problem. In 2016, 10.4 million cases of TB were documented and 10% of this was in children younger than 18 years.1 In Mexico, a toll of 19,812 cases were reported in 2017 in general population, with an incidence rate of 17/100,000 inhabitants.2 Osteoarticular TB (OATB) represents 1% of the extrapulmonary TB in Mexico; however, rib cage TB has been documented only in few cases.1 Usually, the rib cage TB is bereft of clear and specific clinical manifestations, which makes its timely diagnosis difficult. In view of this, it is advisable to suspect this modality of TB in cases involving unspecific signs and symptoms in the chest cage. We report three cases of TB of the ribs in a third-level center in Mexico City.

REPORT OF CASES

Case 1.

A 1-year-old male patient presented heart rates of 111 beats per minute, respiratory frequency of 30 breaths per minute, arterial blood pressure of 90/50 mmHg, temperature 36.9°C, body weight 10.7 kg (percentile 10), and size 88 cm (percentile 10). Before his assessment, he was attended by a private physician who requested only imaging studies, on the basis of which he was referred to the National Institute of Pediatrics for specialized attention. The patient, previously healthy and with Bacillus Calmette–Guérin (BCG) administration at birth, was admitted with a swelling of 6 weeks of duration in the anterior left chest wall, without a history of fever, adenopathies, or other symptoms. Physical examination revealed a 4 × 4 cm mass on the rib grid that was painful to the touch. The mass extended deep into the skin with involvement of the subcutaneous tissue. Pediatric oncology department suspected tumor. Blood test was performed and the results were as follows: white blood cell count, 6.9 cell/mm3; neutrophils, 57.2%; hemoglobin count, 11.6 g/dL; and platelet count, 279,000. Ultrasound reported necrotic nodules of approximately 3.1 cm in diameter and multiple echogenic lesions with “snowflakes” images in their center corresponding to an expanding bone lesion. Computed tomography (CT) images show an abscess deeply located into the thoracic cavity and compressing the pleura without lung involvement or pleural effusion (Figure 1) and destruction of the distal portion of the fifth rib. The abscess was drained obtaining 15 mL of purulent exudate with negative microbiological cultivation and histomorphological report of chronic granulomatous inflammation with Langerhans-type giant multinucleated cells and caseous necrosis. Ziehl–Neelsen stain demonstrated acid-fast bacilli suggestive of TB infection. The patient had no epidemiologically relevant contact, but positive tuberculin skin test (15 × 14 mm). Diagnosis of rib cage TB infection by histopathological studies was reached 3 months after the onset of the disease. Antituberculosis therapy was started with isoniazid, rifampicin, pyrazinamide, ethambutol, and clarithromycin because BCG vaccine (Tokyo 172 sub-strain) infection was suspected. After 2 months of intensive phase treatment, in a continuation phase, the patient was medicated with isoniazid, rifampicin, clarithromycin, and ciprofloxacin because of persistence of purulent exudate. A total of 10 months of treatment and surveillance during 12 months were given. During the second month after starting the treatment, a spontaneous drainage of the lesion occurred, and this persisted until the third month. Healing of the lesion, progressive decrease in its size, and complete disappearance of the same was observed in the sixth month. He was given a monthly follow-up appointment for the next 6 months. The follow-up was clinical without imaging studies or complementary text. On finishing his treatment, his evolution was monitored on outpatient basis through appointments every 3 months. He was finally discharged without data of active sickness, and only the presence of a depressed scar of 3 cm in diameter on the site of lesion could be observed.

Figure 1.
Figure 1.

Case 1: An ostempyesis that introduces into the thorax with compression of the pleura without lung involvement or pleural effusion can be observed (A). In the reconstruction, the destruction of the final portion of the fifth costal arc can be observed (B).

Citation: The American Journal of Tropical Medicine and Hygiene 101, 5; 10.4269/ajtmh.19-0374

Case 2.

A 2-year 7-month-old female patient presented heart rates of 118 beats per minute, respiratory frequency of 26 breaths per minute, arterial blood pressure of 100/63 mmHg, temperature 36°C, body weight 14.8 kg (percentile 75), and size 90 cm (percentile 50). Her arrival to our institute was due to a sickness with 1 week of evolution characterized by an increase in the volume of the left anterior chest, erythema, and hyperthermia, which is accompanied by fever. She was assessed by a physician who started treatment with amoxicillin/clavulanate without improvement on the seventh day after starting the treatment. Ultrasound studies of the lesion was requested and puncture of the same was performed, obtaining purulent material. She was sent to our institute to continue her antibiotic treatment and to assess the extension of the infectious process. On admission, studies, already mentioned in the summary sent, were performed and based on the results a definitive diagnosis was made three weeks after disease onset.

The patient with a history of BCG vaccine administration at birth was attended at emergency department for presenting swelling, erythema, and hyperthermia on the left anterior chest and 1 week of fever duration. Ultrasound performed at the seventh day of treatment with amoxicillin/clavulanate, without improvement, reported an abscess with drainage of purulent exudate.

Blood screening reported leukocyte count of 9,700 cell/mm3, neutrophils of 47%, and platelets of 266,000. Ultrasound revealed a 3.7 × 1.9 cc abscess with necrotic lesions. A 5-day treatment based on dicloxacillin was started but was interrupted because of internal mammary lymphatic growth. A CT scan was performed with a report of a mass on the left chest wall with lytic bony lesions on the 10th rib (Figure 2A) and an underlying abscess extending to the pleura and lung (Figure 2B). Tissue samples reported chronic xanthogranulomatous inflammation with acid-fast bacilli. Treatment based on isoniazid, rifampicin, pyrazinamide, and ethambutol was administered for 12 months. Clinical follow-up and tomographic studies were performed. The persistence of the lesion prevailed until the sixth month with progressive decrease until its resolution. After finishing her treatment, she was monitored through quarterly appointment for 2 years and then finally discharged without complications.

Figure 2.
Figure 2.

Case 2: Reconstruction in 3D with partial destruction of the left 10th costal arc in the final portion (A). Computed tomography scan with the presence of mass dependent on the left costal grid, presence of lytic lesions, and liquid collection with infiltration of lung and pleural tissue (B).

Citation: The American Journal of Tropical Medicine and Hygiene 101, 5; 10.4269/ajtmh.19-0374

Case 3.

A female patient that is 1-year and 5-months-old presented heart rates of 128 beats per minute, respiratory frequency of 32 breaths per minute, arterial blood pressure of 95/56 mmHg, temperature 36.5°C, body weight 11.45 kg (percentile 75), and size 74 cm (percentile 5). Diagnosis was made by histopathological studies on the second month of disease onset.

The patient with a vaccination record of BCG at birth was brought to the hospital for presenting a swelling of 2 months duration on the left hemithorax at the level of the fourth rib, with a size of approximately 9 cm.2 The surrounding skin was indurated and without erythema or local hyperthermia. White blood cell count showed 11.2 cells/mm3 and neutrophils of 33%. The anteroposterior view of a chest X-ray revealed a lytic lesion simulating an expansive bony lesion on the fifth rib that measured 200 mm2 (Figure 3A). Further imaging studies included a CT scan that displayed a lytic lesion on the anterolateral side of 3 cm in length and 1 cm in width (Figure 3B and C). Histopathological review of tissue samples reported chronic xanthogranulomatous inflammation and the presence of Langerhans giant cells along with caseous tissue. As conventional cultures yielded negative results, the diagnosis for a probable case of TB was reached, and treatment with isoniazid, rifampicin, pyrazinamide, and ethambutol was started and continued for 9 months with a favorable clinical picture. Monthly clinical follow-up and tomographic studies during her treatment were performed with the observation of lesion disappearance in the fifth month in control tomography. She was discharged without data of active disease 18 months after finishing her treatment based on favorable response.

Figure 3.
Figure 3.

Case 3: Chest X-ray with a lytic lesion simulating an expansive bony lesion (A) on the fifth rib measuring 200 mm2. Computed tomography scan with a bone lytic lesion on the anterolateral side of the same rib of 3 cm in length (B) and 1 cm in width (C).

Citation: The American Journal of Tropical Medicine and Hygiene 101, 5; 10.4269/ajtmh.19-0374

Information about demographic and clinical information of the presented cases are summarized in Table 1.

Table 1

Clinical and demographic characteristics of presented cases with chest wall tuberculosis without pulmonary involvement

Case 1Case 2Case 3
GenderMaleFemaleFemale
Age of diagnosis1 year2 years and 7 months1 year and 5 months
FeverNoYesNo
Loss of weightNoNoNo
Application of Bacillus Calmette–GuérinYesYesYes
Epidemiological contactNoNoNo
PPD15 × 14 mm5 × 5 mmNo reactive
BAAR in gastric juiceNegativeNegativeNegative
CultureNegativeNegativeNegative
HistopathologyChronic granulomatous inflammation with Langerhans-type giant multinucleated cells and caseous necrosis. Ziehl–Neelsen stain demonstrated acid-fast bacilliChronic xanthogranulomatous inflammation with acid-fast bacilliChronic xanthogranulomatous inflammation and the presence of Langerhans giant cells along with caseous tissue
Time before diagnosis3 months3 weeks2 months

PPD = purified peptide derivative; BAAR = acid-alcohol resistant bacillus.

DISCUSSION

Osteoarticular TB is a diagnostic challenge, which has been described in few studies in pediatric population. Mycobacterium tuberculosis can affect any part of the body from the earliest stage. The most frequent location is the lung, which is the entry route of the bacilli and represents up to 80–85% of the cases of TB in the general population. In Mexico, the national incidence rate of extrapulmonary TB is 2.8/100,000 inhabitants, representing 46.8% of the cases with this type of TB modality. Of these, OATB corresponds to 1–3%.2 Chest wall TB is an unusual form, which occurs in 0.1–1% of OATB cases.3,4

The proposed mechanism of the infection is by hematogenous or lymphatic spread, associated with activation of latent TB, although direct extension has been proposed as another likely mechanism.5 The OATB dissemination is favored by certain factors such as epiphyseal or metaphyseal condition of the weight-bearing bones or the bones that have increased blood flow and bone marrow with abundant lymphoid cells and relatively scarce macrophages. In view of this, the spine is the most affected, followed by hip, knees, and foot bones. Bone erosion results from granulomatous reaction and necrosis, as well as direct invasion of the microorganism.5 The presence of “cold abscesses” is due to the presence of caseous exudate from the regional lymph nodes, explaining the presence of swelling on the physical examination without erythema or hyperthermia.

In our country, the BCG vaccine Tokyo 172 administration is a part of the national immunization scheme, applied at birth, so it is important to consider osteitis due to vaccination as a probable etiologic agent, as reported in some series.6,7 In contrast to other types of TB, which are more common in children aged 2–10 years, the incidence of rib TB is twice higher in men and women of 30 years old. Groups identified as vulnerable are the indigenous population, migrants, rural dwellers, children younger than 5 years, and immunodeficient patients among others.

Preoperative diagnosis is often complex, so open or guided biopsy remains the gold standard. Based on the anatomical location, the radiological findings are divided into four categories: costovertebral (35%), costochondral (13%), the central part of the rib (61%), and multiple cystic lesions.8

Faure et al.9 mention that there is usually only one rib involved and that the most frequent location is the body of the rib, which coincides with our cases. Lytic lesions with periosteal reaction and expansive characteristics are usually observed in imaging studies and less frequently as blunt destruction or pathological fracture.

The expansive nature orients oncological or pyogenic pathologies as differential diagnoses.10 The usual treatment is similar to the one recommended by the WHO for OATB, which is based on isoniazid, rifampicin, pyrazinamide, and ethambutol for 2 months and followed by isoniazid and rifampicin for 10 months. However, in the first cases of OATB, clarithromycin and ciprofloxacin need to be added for the poor clinical course and in suspected infection with the Tokyo 172 strain of the vaccine.11 Complete surgical resection is a controversial option.10

The most important clinical presentations in this series were swelling and pain, with lytic lesions and a soft tissue mass in image studies simulating oncologic pathologies. In all the cases, primary or acquired immunodeficiency was ruled out. Because none of the cases had positive epidemiological contact, TB was initially not considered, so the delay in diagnosis of the three cases from the onset of symptoms was 4, 1, and 2 months respectively. The diagnosis was made through histomorphological analyses.

Finally, is important to consider that primary tumors of the thoracic wall in pediatric population are very rare, with an incidence of less than 2%. Half of the cases are benign, including osteocondroma, chondroma, and fibrosis dysplasia. Bone tumors represent 4.2% of all tumors in children, including osteosarcoma and Ewing’s sarcoma, the latter presenting affectation of the rib cage in 17%. Although the infectious etiology occurs in less than 5%, it should be considered in all those patients who are accompanied by fever, increase in temperature, and hyperemia in the site of lesion.

CONCLUSION

The findings in our cases illustrate that the diagnosis of chest wall TB should be suspected in all patients from endemic areas who present a destructive rib injury. Then, in endemic areas of TB and in which destructive lesions of rib cage are observed, this etiology should always be considered as a differential.

Acknowledgments:

We thank Cyril Ndidi Nwoye Nnamezie, an expert translator whose native language is English, for his help in preparing this manuscript. We also thank the relatives of the patients who gave their consent to carry out this report. This study was funded by the E022 Program of the National Institute of Pediatrics.

REFERENCES

  • 1.

    Rafiqi K, Yousri B, Arihi M, Bjitro C, Aboumaarouf M, El Andaloussi M, 2013. Unusual locations of osteoarticular tuberculosis in children: a report of 12 cases. Orthop Traumatol Surg Res 99: 347351.

    • Search Google Scholar
    • Export Citation
  • 2.

    National Epidemiological Surveillance System (SINAVE) of Mexico, 2015. Epidemiological Profile of Tuberculosis in Mexico [Perfil Epidemiológico de la Tuberculosis en México]. Health Ministry, SINAVE.

    • Search Google Scholar
    • Export Citation
  • 3.

    Agrawal V, Joshi MK, Jain BK, Mohanty D, Gupta A, 2008. Tuberculotic osteomyelitis of rib-a surgical entity. Interact Cardiovasc Thorac Surg 7: 10281030.

    • Search Google Scholar
    • Export Citation
  • 4.

    Kritsaneepaiboon S, Andres MM, Tatco VR, Lim CC, Concepción ND, 2017. Extrapulmonary involvement in pediatric tuberculosis. Pediatr Radiol 47: 12491259.

    • Search Google Scholar
    • Export Citation
  • 5.

    Grover SB, Jain M, Dumeer S, Sirari N, Bansal M, Badgujar D, 2011. Chest wall tuberculosis—a clinical and imaging experience. Indian J Radiol Imaging 21: 2833.

    • Search Google Scholar
    • Export Citation
  • 6.

    Chiu NC, 2015. Mycobacterium bovis bcg-associated osteomielitis/osteítis, Taiwan. Emerg Infect Dis 21: 539540.

  • 7.

    Terreri MT, Yamada AF, 2008. Osteitis caused by BCG vaccination. Pediatr Radiol 38: 481.

  • 8.

    Lee SH, Abramson SB, 1996. Infections of the musculoskeletal system by M. tuberculosis. Rom WN, Garay SM, eds. Tuberculosis. New York, NY: Little Brown and Company, 635644.

    • Search Google Scholar
    • Export Citation
  • 9.

    Faure E, Souilamas R, Riquet M, 1998. Cold abscess of the chest wall: a surgical entity? Ann Thorac Surg 66: 11741178.

  • 10.

    Chang DS, Rafii M, McGuinness G, 1999. Diagnostic issues in tuberculosis of the ribs with a review of 12 surgically proven cases. Respirology 4: 249253.

    • Search Google Scholar
    • Export Citation
  • 11.

    Perez-Velez C, 2012. Pediatric tuberculosis: new guidelines and recommendations. Curr Opin Pediatr 24: 319328.

Author Notes

Address correspondence to Hugo Juarez Olguin, Laboratorio de Farmacología, Instituto Nacional de Pediatría, Avenida Imán N° 1, 3rd piso Colonia Cuicuilco, Mexico City 04530, Mexico. E-mails: juarezol@yahoo.com or adrianos27@hotmail.com

Disclosure: The study was authorized by the Ethics Committee of National Institute of Pediatrics. All data generated or analyzed during this study are included in this published article. Besides, any additional data/files may be obtained from the corresponding author.

Authors’ addresses: Napoleon González Saldaña, Mercedes Macías Parra, Eduardo Arias de la Garza, Diego Galvis Trujillo, Ana Jocelyn Carmona Vargas, Martin Eduardo Palavicini Rueda, and Jose Ivan Castillo Bejarano, Service of Infectology, Instituto Nacional de Pediatria, Mexico City, Mexico, E-mails: drnagosal@hotmail.com, mermacpar@hotmail.com, lalo_arias@hotmail.com, diego.galvis.trujillo@gmail.com, ajocelinc@hotmail.com, mpalavicinir@hotmail.com, and jicastillobejarano@gmail.com. Sara Solorzano Morales, Service of Radiology, Instituto Nacional de Pediatria, Mexico City, Mexico, E-mail: dra.solorzano@yahoo.com.mx. Hugo Juarez Olguin, Laboratory of Pharmacology, Instituto Nacional de Pediatria, Mexico City, Mexico, E-mail: juarezol@yahoo.com.

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