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A 67-year-old woman with a history of noninsulin-dependent diabetes mellitus with bilateral blindness caused by retinopathy, was brought to the hospital because of asthenia and weight loss which had developed during the last 3 months. She had also experienced tumefaction of the second finger of her left hand and mild pain accompanied by suppuration and hearing loss in her right ear. She was febrile (38.8°C) and slightly bradipsychic. The otologic examination showed a papule in the right pinna and otorrhea (see figure1), and an extremely congested auditory canal with two perforations in the tympanum. Pulmonary, cardiac and abdominal examinations were normal. Her left hand is shown in figure 2, and was painful but cold. A soft painless tumour on the palmar side of the wrist prevented her from flexing the second and third fingers.
Blood tests were normal except for erthrocyte sedimentation rate (108-100 mm in first hour), glucose 20.5 mmol/l, sodium 132 mmol/l, lactate dehydrogenase 216 IU/l and alkaline phosphatase 135 IU/l. Basal arterial gasometry was pO2 64 mmHg, pCO2 38 mmHg. Urinalysis revealed pyuria. She was Mantoux negative. Chest X-ray showed a miliary pattern and scars of tuberculous lesions in both lungs. X-Ray of the left hand showed only enlargement of the soft parts of the second finger.
- What is shown in figure 2 ?
- What is the most probable diagnosis ?
- What diagnostic procedure should be carried out ?
- What is the most appropriate treatment ?
Figure 2 shows tumefaction along the whole of the second finger (‘sausage finger’).
The patient had signs of chronic tenosynovitis of the hand with slight pain and cold erythema and of chronic otitis in which early hearing loss and absence of pain attracted our attention. These findings, together with a miliary pattern on the chest X-ray, are highly suggestive of haematogenous tuberculosis.
When a tuberculous aetiology is suspected, it is always crucial to identify Mycobacterium tuberculosis in a culture for an accurate diagnosis and treatment, because the various mycobacterial species vary considerably in their susceptibility to available antimicrobial agents.1 In this case, we cultured specimens of sputum, bronchial brushing and lavage (transbronchial biopsy could not be performed), but all were negative. The biopsy of the ear lesion revealed an unspecific granuloma where acid-fast bacilli were not seen. Magnetic resonance imaging of the affected hand showed an increased signal intensity on T2-weighted images of the tendon sheath, suggesting peritendinous oedema. This was compatible with tendinitis of the flexors of the carpus. An electromyogram, performed to investigate the reason for her inability to flex her second finger, showed a delayed conduction of the median nerve. A surgical decompression was performed. The synovial tissue obtained revealed an important necrotising granulomatous reaction with acid-fast bacilli. Eight weeks later, a specimen of the otic exudate and the synovial tissue grew Mycobacterium tuberculosis, sensitive to the drugs administered to the patient. The remaining cultures, including urine, were negative.
The treatment with tuberculostatics in both otitis and tenosynovitis, does not differ from that of tuberculosis localised elsewhere.1-4 Surgery is recommended to obtain biosy samples when sufficient tissue is affected, or if nerve trapping is suspected.5
Disseminated haematogenous tuberculosis is still a frequent disease nowadays although some characteristics of its presentation have changed. Thus, excluding patients infected by HIV, it has become a disease of middle and advanced ages with high mortality reaching up to 16–28% of cases.6
Tuberculosis used to be the most frequent cause of chronic tenosynovitis of the hand, but today it is rare, causing a delay in diagnosis. Its main features are listed in box FB1. In some cases the surgical debridement may resolve it, but relapses are frequent. It may present as an isolated case or be accompanied by infection in other locations, usually the lung. The presence of a traumatic antecedent which would favour the local activation of a quiescent tuberculous focus, or the setting of the bacillus during haematogenous dissemination has been widely discussed as a predisposing factor for skeletal and muscular tuberculosis.7 If there is no bone involvement, the X-ray shows only enlargement of the soft tissues. The presence of granulomatous tissue obtained by aspiration, open biopsy and complete tenosynovectomy is highly suspicious and may justify starting treatment in anticipation of the results of the cultures.8 The presence of acid-fast bacilli reduces the diagnostic possibilities, but only the presence ofMycobacterium tuberculosis in a culture provides a definitive diagnosis, as other infectious (fungus,Brucella melitensis, nontuberculous mycobacteria) and noninfectious (sarcoidosis, reaction to foreign bodies, inflammatory bowel disease) pathologies can also cause granulomatous tenosynovitis.5
Tuberculous otitis is also an unusual disease nowadays in developed countries, although it used to be a common problem in children.1 The site of entry could be the eustachian tube, but others consider that any caseous focus (mainly pulmonary) may be the source from which bacilli enter the blood stream to reach the temporal bone.3 The suspicious signs and symptoms are listed in box FB2.1 3 4
In conclusion, chronic otitis and tenosynovitis are unusual manifestations of infection by Mycobacterium tuberculosis nowadays. Its nonspecific appearance contributed to a late diagnosis in our patient. A chronic and insidious course, a lack of response to usual treatments and the presence of granulation tissue, should make us aware of this aetiology, so that treatment can be initiated as soon as possible.
Chronic otitis and tenosynovitis for miliary tuberculosis.