Abstract
Rickets is increasingly encountered in practice, even in the Western world. One recent publication reports an overall incidence of 7.5 per 100 000 children. Respiratory infections are well known to be associated with rickets. We present a case of rickets with severe respiratory complications. Tracheobronchomalacia was detected on bronchoscopy. We believe that tracheobronchomalacia may be a hitherto unrecognised finding in rickets and may need to be investigated further.
BACKGROUND
Rickets is being increasingly encountered in practice, even in the Western world. One recent publication reports an overall incidence of 7.5 per 100 000 children.1 The varying clinical presentations are well reported in several published case reports and papers.2,3 In infants and small children respiratory features of vitamin D deficiency described are recurrent infections, stridor and atelectasis.4 Studies in developing countries have also found an association between nutritional rickets and pneumonia.5–7 The cause of this has been related to the action of 1,25-dihdroxycholecalciferol on the immune system.8,9 However, there is little information on the pathophysiology of respiratory complications.
The possible association between tracheobronchomalacia and rickets has not been described before.
CASE PRESENTATION
A 7-month-old child presented with acute onset wheeze, and rapid deterioration suggestive of a viral-induced wheeze/bronchiolitis. At admission, she was noted to have a mild pallor, in addition to the features of rickets including Harrison’s sulcus, rickety rosary and widened wrists, which were confirmed on radiology (fig 1) and on biochemistry results.
Figure 1.
Bony changes before and after treatment of rickets.
The baby was born a term baby with a birth weight of 1.4 kg, secondary to maternal pre-eclampsia. At admission, her weight was 5 kg (<0.4 centile), length 60 cm (<0.4 centile) and head circumference 44 cm (2nd centile). She was developmentally normal except for a mild motor delay. A dietetic history revealed that she was exclusively breast fed till 6 months of age and was on a weaning diet of porridge, butternut squash, root vegetables, chicken and fruits. Her rickets was thought to be because of poor sun exposure in the mother, as she had a photosensitive skin condition.
After admission, the child’s condition deteriorated and she needed ventilatory support for a few days. Even though she improved, she had two further similar episodes. On the third intensive care admission, flexible bronchoscopy was performed which revealed a diffuse bronchomalacia mainly affecting the right main bronchus. In view of this finding, long-term non-invasive ventilation was initiated.
INVESTIGATIONS
The investigations carried out are shown in table 1.
Table 1.
Summary of key investigations
| Test | At admission | At 3 months |
| Calcium | 1.59 mmol/l (ionised 0.9) | 2.4 mmol/l |
| Corrected calcium | 1.63 mmol/l | 2.63 mmol/l |
| Phosphate | 1.0 mmol/l | 2.1 mmol/l |
| Albumin | 43 g/l | 44 g/l |
| Alkaline phosphatase | 2293 IU/l | 563 IU/l |
| Magnesium | 0.75 mmol/l | 0.91 mmol/l |
| 25-Hydroxy vitamin D | <6 nmol/l | 67 nmol/l |
| Parathyroid hormone | 464 ng/l | |
| Thyroid-stimulating hormone | 2.99 mIU/l | |
| Haemoglobin | 6.1 g/dl | 12.6 g/dl |
TREATMENT
Treatment for rickets and nutritional rehabilitation in addition to non-invasive ventilation.
OUTCOME AND FOLLOW-UP
The child had a repeat bronchoscopy in 3 months which showed a marked improvement in the bronchomalacia. Her mother was advised to discontinue the non-invasive ventilation.
At 13 months of age, she was 7.2 kg (just below the 0.4th centile), on the 9th centile for length and the 25th centile for head circumference. She did not have clinical or radiological signs of rickets.
DISCUSSION
This case was at the severe end of the spectrum of rickets, with recurrent respiratory complications requiring bronchoscopy. This revealed the findings of bronchomalacia in the presence of rickets.
The presence of tracheobronchomalacia is usually seen with laryngomalacia or cardiac defects,10,11 but has not been reported with rickets before. This possible association could explain the increased incidence of pneumonia and, to a certain extent, the mortality and morbidity in acute respiratory infections in children with severe rickets. The role of early institution of respiratory support to improve patient outcome and prevent recurrent respiratory collapse in severe cases such as these needs to be further looked into. Issues to highlight here are that early bronchoscopy, if available, may obviate further respiratory complications. Bronchomalacia following severe rickets may improve following combination treatment such as dietary and respiratory support. Severe rickets may account for this degree of bronchomalacia in such children without other risk factors such as prematurity or cardiac defects.
LEARNING POINTS
Rickets is known to be associated with recurrent respiratory infections.
Tracheobronchomalacia may be associated with respiratory morbidity in rickets.
Further studies may be useful to look at this possible association.
Footnotes
Competing interests: none.
Patient consent: Patient/guardian consent was obtained for publication.
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