Open Access

Cystic tuberculosis of the scapula in a young boy: a case report and review of the literature

  • Deepali Jain1,
  • Vijay K Jain2Email author,
  • Yashwant Singh2,
  • Satish Kumar1 and
  • Deepak Mittal1
Journal of Medical Case Reports20093:7412

DOI: 10.4076/1752-1947-3-7412

Received: 5 September 2008

Accepted: 15 July 2009

Published: 5 August 2009

Abstract

Introduction

Tuberculosis of the flat bones is rare and only a small percentage involves the scapular bone.

Case presentation

We report a rare case of tuberculosis of the scapula in a 14-year-old. Diagnostic clues include lytic areas with low density seen in the body of the scapula involving a glenoid margin associated with typical clinical features. Treatment should include a regimen of four antitubercular drugs along with surgical debridement if required.

Conclusion

Although rare, tuberculosis should be suspected in patients presenting with a chronic sinus in the scapular region, particularly in the developing world.

Introduction

Tuberculosis (TB) has been a health concern for several thousand years. Only a small number of patients with tuberculosis will have osteoarticular involvement [1]. Less than one percent of all osteoarticular TB affects the shoulder, a fraction of it involving the scapular bone itself [2]. To the best of our knowledge, only eleven cases of scapular tuberculosis have been reported to date [3]-[12]. We present the 12th case, occurring in a pediatric patient, which has been described only twice before in the English literature ([Table 1]) [5, 10].
Table 1

Review of the literature of previously reported cases of TB of the scapula

S.N

Author year

No. of patients

Age/sex

Location

Side

Presenting complaints

Other sites

Treatment

1

Lafond 1958 [3]

One

NA

NA

NA

NA

NA

NA

2

Martini et al. 1986 [4]

One

NA

Acromian

NA

NA

NA

NA

3

Shannon et al. 1990 [5]

One

4/male

Scapula

Lt

Pain and swelling of the left shoulder

Isolated with Rt ileum involvement, multifocal cystic

ATD

4

Mohan et al. 1991 [6]

One

23/female

Body of scapula

Rt

Pain and swelling

Isolated

Drainage and ATD

5

Gusati et al. 1997 [7]

One

NA

Spine of scapula

NA

Pain

Isolated

Surgery and ATD

6

Vohra et al. 1997 [8]

One

NA

Body of scapula

NA

NA

Isolated

NA

7

Kam et al. 2000 [9]

Two

31/male 22/female

Acromian, Lareral border of scapula

Rt Rt

1) Pain and swelling 2) Incidental finding

Isolated, Multifocal (T12 and L2 vertebrae; upper part of the right sacroiliac Joint)

Debridement and curettage and ATD, ATD alone

8

Greenhow and Weintrub 2004 [10]

One

14/female

Inferior aspect of the left scapula

Lt

Enlarging, nontender mass

Cystic lesion with a soft tissue component, located dorsal to the left scapula

Scapular mass excision

9

Stones and Schoeman 2004 [11]

One

42/male

Scapula

NA

Discharging sinus

As apart of multimodal tuberculosis involving maxilla, parital bones and spine

Died

10

Husen et al. 2006 [12]

One

18/male

Spine of scapula near neck

Lt

Diffuse pain

Isolated

ATD

11

Present case 2007

One

14/male

Body of scapula involving glenoid margin

Rt

Pain swelling and discharging sinus

Isolated

ATD

Abbreviations: Rt, right; Lt, left; NA, not available; ATD, anti-tubercular drugs.

Case presentation

A 14-year-old boy, from a low socio economic background presented with a four-month history of pain, and a discharging sinus in the right upper scapular region that had been present for two months. The pain had been gradual, dull and aching. The patient had been treated for these complaints without relief and had developed a scapular swelling which broke down and discharged serosanguinous fluid. He had an antecedent history of trauma and an associated history of fever, weight loss, loss of appetite, night sweats, malaise and fatigue. He had no history of previous pulmonary or extrapulmonary tuberculosis and there was no family history of tuberculosis.

On local examination, we observed a sinus measuring less than 1 cm in size overlying the right upper scapular region. It was slightly tender, adherent to the bone and surrounding soft tissue, with associated granulation tissue and serosanguinous discharge and the surrounding skin was indurated and unhealthy. There was no significant regional lymphadenopathy, he had a full range of motion of the shoulder joint and there was no tenderness over the spine and paraspinal muscles in the thoracic region. Laboratory examination showed only a minimally increased white blood cell count (10950/mm3) with a predominance of lymphocytes (48%), elevated erythrocyte sedimentation rate (ESR) of 65 mm (Westergren method) after one hour and a positive C-reactive protein (CRP) test. A Mantoux tuberculin skin test (purified protein derivative, five tuberculin units) was positive with 15 mm of induration observed 48 hours after administration. Anteroposterior radiographs of the right shoulder showed two rounded oval lytic areas with low density seen in the body of the scapula involving the glenoid margin (Figure 1) and there was a minimal increase in density surrounding the lesion. A plain chest radiograph was normal and a closed core biopsy of the sinus tract revealed epithelioid cell granulomas with central necrosis, typical Langhans giant cells and a positive stain for acid fast bacilli by Ziehl-Neelsen stain (Figure 2). On microbiologic examination positive culture on Lowenstein-Jenson medium for AFB was present. Anti-tuberculosis chemotherapy began immediately. The patient received four months of anti-tubercular chemotherapy, consisting of four drugs: isoniazid (INH), pyrazinamide, ethambutol and rifampicin. He was given INH, rifampicin and ethambutol for four months and INH and rifampicin for 10 months. Radiographs at 10 months showed complete resolution of the bony lesion. The sinus healed without any complications after four months of anti-tubercular treatment. The patient's appetite improved, he gained weight and his growth indices significantly improved at the end of the anti-tubercular treatment. At two-year follow-up he was asymptomatic.
Figure 1

Anteroposterior (AP) radiograph of the shoulder showing two well defined lytic destructive lesions involving the glenoid margin suggestive of cystic tuberculosis.

Figure 2

Microphotograph showing epithelioid cell granulomas with necrosis. H&E ×40.

Discussion

Osteoarticular tuberculosis accounts for 3% of all cases of tuberculosis and isolated tuberculosis of the scapula is rare. In past reports most cases were associated with other forms of tubercular osteomyelitis and only six were isolated to the scapula [5]-[9, 12]. We report tuberculosis of the scapula in a 14-year-old male patient. Previously, Greenhow and Weintrub [10] also reported tubercular involvement of the scapula in a pediatric patient. Clinically, patients with osteoarticular tuberculosis present with localized symptoms of swelling and pain as was present in our case. Radiograph of the shoulder showed a well defined lytic destructive lesion of the scapula indicative of cystic tuberculosis. Cystic tuberculosis is a rare form of tuberculosis seen mostly in children and young adults, usually in the appendicular skeleton; occasionally involving flat bones as seen in the present case. Cystic tubercular involvement of the scapula has only once been reported, in the literature [5] and there seems to be a changing pattern of cyst-like lesions in osseous tuberculosis. Multicystic and multifocal lesions were more common 50 years ago, but it seems that solitary lesions are now predominant and this may be related to immunological factors. Vohra et al. [8] detected nine solitary cystic lesions in six adults and three children. In the present case we found two cystic lesions near the glenoid margin of the scapula. Bone lesions were usually solitary because of sensitization of the patient to the tubercle bacillus; however, if host immunity is poor and the immune response has been altered, the lesions may multiply. Trauma probably draws the attention to a mild focus or it may activate a latent tubercular focus. Sinus formation and abscess are common in tuberculous osteitis as seen in our case. The diagnosis of tuberculosis was based on the staining of smears for acid-fast bacilli and culturing for mycobacteria. AFB smear results lack sensitivity and are not specific for tuberculosis [13] and while mycobacterial culture and identification is specific for diagnosis, it takes two to three weeks. Histologic diagnosis in conjunction with microbiologic and molecular testing should be considered appropriate for the diagnosis.

Conclusion

Although rare, tuberculosis should be suspected in patients presenting with a chronic sinus in the scapular region, particularly in the developing world. As uncommon presentations and sites of osteoarticular disease can be a source of delay and error in management, an open biopsy may be necessary in doubtful cases.

Consent

Written informed consent was obtained from the patient's parent for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Abbreviation

TB: 

tuberculosis.

Declarations

Authors’ Affiliations

(1)
Department of Pathology, Maulana Azad Medical College
(2)
Departments of Orthopaedics & Radiodiagnosis, Dr. Ram Manohar Lohia Hospital

References

  1. Morris BS, Varma R, Garg A, Awasthi M, Maheshwari M: Multifocal musculoskeletal tuberculosis in children: appearances on computed tomography. Skeletal Radiol. 2002, 31: 1-8. 10.1007/s00256-001-0439-y.View ArticlePubMedGoogle Scholar
  2. Batman JE: Shoulder and neck. WB Saunders & Co. Philadelphia 1978Google Scholar
  3. Lafond EM: An analysis of adult skeletal tuberculosis. J Bone Joint Surg Am. 1958, 40: 346-364.PubMedGoogle Scholar
  4. Martini M, Adjrad A, Boudjemaa A: Tuberculous osteomyelitis. A review of 125 cases. Int Orthop. 1986, 10: 201-207.PubMedGoogle Scholar
  5. Shannon FB, Moore M, Houkom JA, Waecker NJ: Multifocal cystic tuberculosis of bone. Report of a case. J Bone Joint Surg Am. 1990, 72: 1089-92.PubMedGoogle Scholar
  6. Mohan V, Danielsson L, Hosni G, Gupta RP: A case of tuberculosis of the scapula. Acta Orthop Scand. 1991, 62: 79-80. 10.3109/17453679108993101.View ArticlePubMedGoogle Scholar
  7. Guasti D, Devoti D, Affanni M: Tubercular osteitis of the spine of the scapula. Chir Organi Mov. 1997, 82: 413-418.PubMedGoogle Scholar
  8. Vohra R, Kang HS, Dogra S, Saggar RR, Sharma R: Tuberculous osteomyelitis. J Bone Joint Surg [Br]. 1997, 79: 562-566. 10.1302/0301-620X.79B4.7618.View ArticleGoogle Scholar
  9. Kam WL, Leung YF, Chung OM, Wai YL: Tuberculous osteomyelitis of the scapula. Int Orthop. 2000, 24: 301-302. 10.1007/s002640000165.View ArticlePubMedPubMed CentralGoogle Scholar
  10. Greenhow TL, Weintrub PS: Scapular mass in an adolescent. Pediatr Infect Dis J. 2004, 23: 84-85. 10.1097/01.inf.0000105323.67815.d5.View ArticlePubMedGoogle Scholar
  11. Stones DK, Schoeman CJ: Calvarial tuberculosis. J Trop Pediatr. 2004, 50: 361-364. 10.1093/tropej/50.6.361.View ArticlePubMedGoogle Scholar
  12. Husen YA, Nadeem N, Aslam F, Shah MA: Tuberculosis of the scapula. J Pak Med Assoc. 2006, 56: 336-338.PubMedGoogle Scholar
  13. Tenover FC, Crawford JT, Huebner RE, Geiter LJ, Horsburgh CR, Good RC: The resurgence of tuberculosis: is your laboratory ready?. J Clin Microbiol. 1993, 31: 767-770.PubMedPubMed CentralGoogle Scholar

Copyright

© Jain et al.; licensee Cases Network Ltd. 2009

This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.