A thirty-four-day-old female infant was brought by her mum for consultation presenting with swollen and painful left upper and lower limbs since 1 month. Her mum initially noticed swelling of the left knee and leg with reduced movement on day 6 of life for which she returned with baby to the hospital. The consulting specialist attributed this to an infiltration or localized inflammation as a result of blood sample collection for Bilirubinemia done on day 2 of life for the baby. Alcohol dressing was done on the affected limb and mum returned home with baby. Mum brought baby back two days later, due to the persistence of swelling and now reduced movements of left upper limb. The baby had fever at 37.8 degrees and was given Paracetamol syrup, dose for age. An X-ray of limb was done which was reported as normal. Physiotherapy sessions were prescribed and started that same day, but few days later, mum noticed the left upper limb had become less mobile as well. Physiotherapy sessions were continued with very little change. The onset of a swelling on the right thumb with a small circumscribed collection led to another consultation in the same health center few days later. Persistence of symptoms after about a week led to the present consultation in our health structure.
This baby was born by elective cesarean section indicated for eclampsia in mum. Pregnancy had been averagely well-followed up.Mum’s blood pressure was stable all through the pregnancy and routine infectious work-ups were negative. Mum’s Hb electrophoresis is AA and blood group O+. She doesn’t have any known chronic diseases apart from eclampsia during this pregnancy. There is no known chronic disease in the father or two siblings.
On clinical exam, the infant is calm and vital signs are stable. There was shortening of left lower limb with swelling, warmth and tenderness. Reduced motion of left upper limb with normal movement of fingers.
STOP. THINK. DIAGNOSE.
Diagnosis was Chronic septic arthritis on a terrain to be investigated. First differential Chronic osteomyelitis.
Probable terrains were HIV, hemoglobinopathy, Diabetes or malformation of blood vessels.
The pediatric surgeons were contacted for surgical opinion.
Work-ups requested were X-rays of left upper and lower limbs, soft tissue ultrasound of left knee and shoulder joints, blood cultures, culture of liquid to be aspirated from left knee, HIV serology and Hemoglobin electrophoresis. Full Blood Count and CRP were also requested.
X-rays were in favour of old bone lesions indicating advanced osteomyelitis. FBC with moderate normocytic normochromic anemia with Hb:11.8g/dL, CRP:38.8mg/l but all cultures were negative. HIV serology negative. Hemoglobin electrophoresis result suggested delta-beta thalassemia, with the absence of A2 hemoglobin and predominance of HbF.
Patient was managed with IV empirical antibiotics (Ceftriaxone, Cloxacillin at high doses for 10 days, Gentamicin for 3 days). The left limbs were immobilized in casts on day 5 of admission.
On day 10 of hospitalization, there was no more swelling and warmth of affected limbs, right thumb lesion had dried up, CRP was negative, and patient was discharged on oxacillin syrup therapeutic dose for two weeks. Follow-up is to continue every week with the team. Hb electrophoresis would be done again at 3 months of life to confirm Hb pattern.
Delta thalassemia is usually asymptomatic or with just mild symptoms like of anemia. A beta thalassemia could be masked by the presence of an associated delta thalassemia, but then one would expect the reduced presence of Hb A2 rather than total absence. Also worth pondering about, if a bone infection can be directly linked to the terrain of hemoglobinopathy. We await your contributions for this case and Hb electrophoresis result at 3 months.
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