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Congenital pseudoarthrosis of the tibia with localised gigantism in a case of congenital constriction band syndrome.

Agashe MV, Rathod CM, Dhamele JA - Indian J Plast Surg (2011)

View Article: PubMed Central - PubMed

Affiliation: Department of Paediatric Orthopaedics, Bai Jerbai Wadia Hospital for Children, Mumbai, India.

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He had progressively increasing volume of the left lower limb and no movements of the ankle and toes... The child was born normally and there was no adverse perinatal history... The right lower limb showed superficial constriction bands over the foot and calf, with minimal distal swelling... Radiograph of the left lower limb showed a cyst-like lesion in the distal tibia with a pathological fracture similar to type III of Boyd's classification for congenital pseudoarthrosis of the tibia... The patient had an uneventful post-operative recovery and all surgical wounds healed well [Figure 4]... A decision regarding ambulation with a prosthesis would be taken later... Amniotic constriction band syndrome is a group of disorders characterised by a wide range of congenital anomalies, including annular constrictions over the extremities, acrosyndactyly, talipes equinovarus, cleft lip and palate and hemangiomas... According to Paterson's classification of constriction band syndromes, our patient seems to be in type II, with distal lymphedema and sensory deficits... Closed pseudoarthrosis of the tibia, which has been noted earlier in congenital amniotic band syndrome, usually requires no treatment other than adequate splintage... However, this was not possible here due to the associated severe anomalies... Neurocirculatory disturbance along with lymphatic obstruction of such magnitude has not been described before, although a similar case of neurodeficit without circulatory disturbances has been described earlier by Nambi et al... In conclusion, this case is presented to discuss the end result of untreated congenital constriction band syndrome as well as to present this unique combination of severe lymphedema leading to localised gigantism and congenital pseudoarthrosis of the tibia in the setting of Streeter's dysplasia.

No MeSH data available.


Related in: MedlinePlus

Clinical photograph of the left leg and foot showing severe swelling with nubbins visible. There was no discernible ankle and foot
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Figure 2: Clinical photograph of the left leg and foot showing severe swelling with nubbins visible. There was no discernible ankle and foot

Mentions: A 6-month-old male child was brought to our institute for deformity in both hands and feet noticed by the parents since birth. He had progressively increasing volume of the left lower limb and no movements of the ankle and toes. The child was born normally and there was no adverse perinatal history. The left lower limb showed a deep, circumferential constriction band near the lower-third of the leg with abnormal mobility at the site with gross enlargement distally. A few nubbins could be felt instead of the toes. No movements could be elicited from the ankle [Figures 1 and 2]. The limb was insensate distal to the constriction band. Pulsations of the dorsalis pedis and the posterior tibial artery could not be palpated. There were constriction bands in the other limbs without any concomitant swelling with acrosyndactylous digits. The right lower limb showed superficial constriction bands over the foot and calf, with minimal distal swelling. The right lower limb had normal sensations and movements. Radiograph of the left lower limb showed a cyst-like lesion in the distal tibia with a pathological fracture similar to type III of Boyd's classification[1] for congenital pseudoarthrosis of the tibia. Radiograph of the right lower limb was normal [Figures 3a and b].


Congenital pseudoarthrosis of the tibia with localised gigantism in a case of congenital constriction band syndrome.

Agashe MV, Rathod CM, Dhamele JA - Indian J Plast Surg (2011)

Clinical photograph of the left leg and foot showing severe swelling with nubbins visible. There was no discernible ankle and foot
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3111106&req=5

Figure 2: Clinical photograph of the left leg and foot showing severe swelling with nubbins visible. There was no discernible ankle and foot
Mentions: A 6-month-old male child was brought to our institute for deformity in both hands and feet noticed by the parents since birth. He had progressively increasing volume of the left lower limb and no movements of the ankle and toes. The child was born normally and there was no adverse perinatal history. The left lower limb showed a deep, circumferential constriction band near the lower-third of the leg with abnormal mobility at the site with gross enlargement distally. A few nubbins could be felt instead of the toes. No movements could be elicited from the ankle [Figures 1 and 2]. The limb was insensate distal to the constriction band. Pulsations of the dorsalis pedis and the posterior tibial artery could not be palpated. There were constriction bands in the other limbs without any concomitant swelling with acrosyndactylous digits. The right lower limb showed superficial constriction bands over the foot and calf, with minimal distal swelling. The right lower limb had normal sensations and movements. Radiograph of the left lower limb showed a cyst-like lesion in the distal tibia with a pathological fracture similar to type III of Boyd's classification[1] for congenital pseudoarthrosis of the tibia. Radiograph of the right lower limb was normal [Figures 3a and b].

View Article: PubMed Central - PubMed

Affiliation: Department of Paediatric Orthopaedics, Bai Jerbai Wadia Hospital for Children, Mumbai, India.

AUTOMATICALLY GENERATED EXCERPT
Please rate it.

He had progressively increasing volume of the left lower limb and no movements of the ankle and toes... The child was born normally and there was no adverse perinatal history... The right lower limb showed superficial constriction bands over the foot and calf, with minimal distal swelling... Radiograph of the left lower limb showed a cyst-like lesion in the distal tibia with a pathological fracture similar to type III of Boyd's classification for congenital pseudoarthrosis of the tibia... The patient had an uneventful post-operative recovery and all surgical wounds healed well [Figure 4]... A decision regarding ambulation with a prosthesis would be taken later... Amniotic constriction band syndrome is a group of disorders characterised by a wide range of congenital anomalies, including annular constrictions over the extremities, acrosyndactyly, talipes equinovarus, cleft lip and palate and hemangiomas... According to Paterson's classification of constriction band syndromes, our patient seems to be in type II, with distal lymphedema and sensory deficits... Closed pseudoarthrosis of the tibia, which has been noted earlier in congenital amniotic band syndrome, usually requires no treatment other than adequate splintage... However, this was not possible here due to the associated severe anomalies... Neurocirculatory disturbance along with lymphatic obstruction of such magnitude has not been described before, although a similar case of neurodeficit without circulatory disturbances has been described earlier by Nambi et al... In conclusion, this case is presented to discuss the end result of untreated congenital constriction band syndrome as well as to present this unique combination of severe lymphedema leading to localised gigantism and congenital pseudoarthrosis of the tibia in the setting of Streeter's dysplasia.

No MeSH data available.


Related in: MedlinePlus