|Year : 2022 | Volume
| Issue : 2 | Page : 128-130
An adolescent girl with lytic erosion of the rib: What is the diagnosis?
Chirag Thakur1, Sachin Singh1, Anmol Bhatia2, Manish Rohilla3, Meenu Singh1, Joseph L Mathew1
1 Department of Pediatrics, PGIMER, Chandigarh, India
2 Department of Radio-Diagnosis and Imaging, PGIMER, Chandigarh, India
3 Department of Cytology and Gynecological Pathology, PGIMER, Chandigarh, India
|Date of Submission||24-Apr-2022|
|Date of Decision||25-Apr-2022|
|Date of Acceptance||26-Apr-2022|
|Date of Web Publication||30-May-2022|
Prof. Joseph L Mathew
Advanced Pediatrics Centre, PGIMER, Chandigarh
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Thakur C, Singh S, Bhatia A, Rohilla M, Singh M, Mathew JL. An adolescent girl with lytic erosion of the rib: What is the diagnosis?. Indian Pediatr Case Rep 2022;2:128-30
|How to cite this URL:|
Thakur C, Singh S, Bhatia A, Rohilla M, Singh M, Mathew JL. An adolescent girl with lytic erosion of the rib: What is the diagnosis?. Indian Pediatr Case Rep [serial online] 2022 [cited 2022 Jul 4];2:128-30. Available from: http://www.ipcares.org/text.asp?2022/2/2/128/346246
We describe the clinical course of an adolescent girl, who presented with unilateral chest pain, and progressively worsening shortness of breath. The clinical examination confirmed the presence of pleural effusion. On radiological evaluation, the lungs were normal, but there was a lytic lesion in the anterior end of a single rib, with periosteal thickening of multiple ribs on the same side. There was associated pleural thickening as well. The learning objectives of this case are to demonstrate a clinicoradiological approach toward the diagnosis and the use of appropriate investigations to confirm the diagnosis.
| Clinical Description|| |
A 12-year-old girl presented with left-sided chest pain and shortness of breath for 8 months. At the onset of her illness, she had sustained a trivial trauma to the left side of the chest. As she walked past a stationary motorcycle, its handle grazed her left chest. Within a few hours, she perceived a dull ache at the site of contact and a pea-sized swelling that was soft but slightly tender to touch. There was no history of any bleeding or bruising of the overlying skin. She was able to breathe normally. A local private practitioner opined that it could be a fracture of a rib, requiring only conservative treatment. However, it was not confirmed with a radiograph. The swelling gradually subsided over the next few days, but the dull pain persisted. Over the next few weeks, the discomfort became more diffuse, extending to the entire left side of her chest. The child reported aggravation on exertion and deep breathing, but it was not severe enough to limit her daily activities. A month later, she developed shortness of breath which was insidious in onset. Initially, it occurred with exertion (like climbing the stairs), but over 3 months, it gradually progressed to being present while walking, although not at rest.
Throughout this period, there was no history of fever, cough, loss of appetite, night sweats, bony aches and pain, increasing pallor, bleeding from any site, recurrent ulcers in the mouth, rash, photosensitivity, and pain or swelling of any joints. The child's weight remained static for about a year. The past history was not significant. There was no history of contact with tuberculosis (TB). She had been living with her grandmother since the age of 8 years. Thus, her details before that were not available. She was an average student at school.
On examination, she was afebrile, with a pulse rate of 88/min (min), respiratory rate of 24/min, SpO2 99% in room air, and blood pressure of 100/62 mmHg. Her weight was 29.5 kg (−1.87 Z score), height 147.5 cm (−0.64 Z score), and body mass index 13.56 kg/m2 (−2.04 Z score) as per the Indian Academy of Pediatrics growth chart reference values. General physical examination did not reveal pallor, icterus, cyanosis, clubbing, lymphadenopathy, rash, or edema. The BCG scar was present. Bone and joint examination was normal. The respiratory system examination revealed a symmetric chest with equal expansion, mild tracheal deviation to the right side, apex beat located 1 cm internal to the midclavicular line in the left fifth intercostal space, stony dullness in the left lower intercostal spaces, and diminished breath sounds in the same locations. There were no audible crackles or wheeze. These clinical findings suggested a left-sided pleural effusion. The examination of the other systems was unremarkable. A chest radiograph was ordered [Figure 1].
|Figure 1: Chest X-ray showing left pleural effusion with collapse consolidation of the underlying lung, with broadening of the anterior end of the eighth ribs on the left side (yellow arrow)|
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What are the salient findings in her chest X-ray?
The radiograph was a well-centered, well-exposed film. The striking finding was a homogeneous opacity in the left lower zone, obliterating the left costophrenic angle, and silhouetting the left heart border and diaphragm. There was no obvious tracheal deviation. These radiological findings confirmed the presence of a left-sided pleural effusion with possible collapse or consolidation (or both) of the underlying lung. Closer examination revealed broadening of the anterior end of the left eighth rib with the suspicion of a lytic lesion within it. The other ribs and soft tissues appeared unremarkable. A left-sided paratracheal lymph node was suspected but could not be confirmed.
What are the clinical possibilities based on the clinical history and radiograph?
We considered the following differential diagnoses based on the clinical phenotype of a suspected rib fracture following trivial trauma, a lytic bony lesion, and slowly progressing ipsilateral pleural effusion: rib bone TB, a malignant bone tumor such as Ewing's sarcoma, a benign bone tumor such as osteochondroma or eosinophilic granuloma. However, the absence of fever went against TB, the progression over months was unusual for Ewing's sarcoma, and the absence of a palpable tumor made the possibility of benign growths less likely.
What should be the next line of investigations?
Further investigations were planned as per the differential diagnoses considered. An ultrasonographic examination of the chest showed a loculated left pleural effusion of 5.2 cm × 2.6 cm. Ultrasonographic-guided pleural tap yielded yellowish fluid with rich cellularity; total leukocyte count was 14,080/mm3 with 99.3% lymphocytes, and no malignant cells. Gram staining, Ziehl − Neelsen (ZN) staining, bacterial culture, GeneXpert, liquid medium mycobacterial culture, and fungal culture were negative. The tuberculin skin test was reactive (25 mm × 26 mm). Three samples of gastric lavage (as per institutional protocol) underwent ZN staining, GeneXpert, and liquid medium mycobacterial culture but were negative. Fiber-optic bronchoscopy was performed and the bronchoalveolar lavage revealed predominant lymphocytes without any malignant cells. ZN stain, GeneXpert, and liquid medium mycobacterial culture did not indicate TB.
What should be the next step in the clinical approach?
As the child had a history of suspected rib fracture following trivial trauma, and the pleural fluid analysis was inconclusive, we planned a contrast-enhanced computed tomography of the thorax [Figure 2].
|Figure 2: Contrast-enhanced computed tomography sections showing: (a) Right paratracheal and para-aortic lymph nodes (red arrows) in the medistinal window; (b) broadening of the ribs on the left side (compared to the right side), suggesting periosteal reaction (yellow arrows); (c) Left-side pleural effusion with underlying collapse consolidaton and (d) Lytic erosion of the rib on the left side (red arrowhead)|
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What are the salient findings in the contrast-enhanced computed tomography thorax images?
The mediastinal window sections showed multiple, enlarged, discrete, and conglomerating lymph nodes in the prevascular, pretracheal, right paratracheal, precarinal, and bilateral hilar regions. There was sheet-like soft-tissue thickening (1.5 cm) along the costal, diaphragmatic, and mediastinal pleura on the left side with nodularity. Solid periosteal thickening involving multiple ribs on the left side and a focal lytic erosion in the anterolateral aspect of the eighth rib were noted. There was moderate pleural effusion with underlying collapse consolidation of the left lung. The radiological diagnosis based on these findings was an underlying lymphoreticular malignancy like leukemia or lymphoma.
How to proceed further?
A pleural fluid flow cytometry analysis was performed which suggested reactive pleural effusion without any evidence of malignant cells. Therefore, an ultrasonography-guided fine-needle aspiration (FNA) of the mediastinal lymph node was done. This showed multi-nucleated giant cells with epithelioid granuloma and the presence of acid-fast bacilli (AFB) on ZN staining. However, GeneXpert and liquid medium mycobacterial culture from the FNA sample were negative. There were no abnormal cells suggestive of lymphoreticular malignancy. Thus, based on these findings, mediastinal lymph node TB was confirmed, and the other sites of involvement (ribs and pleura) were also presumed to be due to the same etiology. In consultation with the orthopedic team, it was decided that a rib bone biopsy would be performed only in the event of nonresponse to anti-TB treatment (ATT).
| Management and Outcome|| |
Standard four-drug ATT was started along with pyridoxine, and the child was discharged. She was closely monitored every 2 weeks to observe clinical improvement. By the end of the intensive phase of ATT, the child was symptomatically well; the chest pain and breathlessness had resolved. During this period, weight gain of 1 kg was recorded. Ultrasonography of the chest showed a reduction in pleural effusion to 4.2 cm × 1.6 cm (around 30 cc volume). Therefore, it was decided to shift to the maintenance phase of ATT with continuation of monthly follow-up.
| Discussion|| |
Osteoarticular TB accounts for only 10%–20% of extrapulmonary TB in children. There are two broad pathophysiologic mechanisms of rib involvement; the first is by lymphatic or hematogenous spread. The second is by the direct extension from contiguous sites such as the lung or pleura. In the index case, either mechanism could be possible, although the presence of mediastinal TB lymphadenopathy suggests a higher likelihood of lymphatic spread from that site. After the initial infection of the bone, there is marked local hyperemia that results in osteoporosis. In the early stages, a radiograph may appear normal, but more advanced cases may show changes ranging from mild osteopenia to lesions suggestive of osteomyelitis. Computed tomography scan can help in identifying the extent of lesions. The radiographic features of bone TB may mimic eosinophilic granuloma, malignancy, or fungal infections. Hence, it is difficult to establish the diagnosis of osteoarticular TB solely on the basis of radiological findings. In addition, <50% patients have concomitant pulmonary involvement. Therefore, tissue diagnosis from the affected bone and/or other sites is ideal to confirm the diagnosis.
There is controversy regarding whether a bone biopsy or surgical removal of the rib lesion is mandatory for establishing the diagnosis of tuberculous osteomyelitis. There are several published case series and case reports, in which the diagnosis of TB could only be established on histopathology after surgical resection of the rib.,, However, in TB endemic areas, due to the invasive nature of the aforementioned approach, ATT is often initiated on presumption and the response to treatment is considered an indirect confirmation of the diagnosis. In this case, the orthopedicians were of the opinion that the removal of the rib would lead to instability of the thoracic cage. They suggested that taking this risk was unjustified, since histopathology of the mediastinal lymph node had already demonstrated the presence of epithelioid cell granulomas, with giant cells and AFB. Hence, the diagnosis remained presumptive. Confirmation by molecular diagnostic methods or culture should always be attempted in all cases since these findings can also occur with infection by nontubercular mycobacteria and nocardia.
There are two views regarding the management of rib TB. Some experts emphasize the need for surgical resection or debridement along with ATT,, whereas others suggest that medical therapy alone for 12 months may suffice, reserving surgical debridement for only the 10%–15% cases that fail to improve with ATT.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2]