Clinical Features and Operative Findings of Congenital Flexion Deformity of Multiple Digits

Ogino, Toshihiko; Ishii, Seiichi; Kato, Hiroyuki

doi:10.1111/j.1741-4520.1993.tb00539.x

Cited by 3 publications

(5 citation statements)

References 12 publications

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“…As for the treatment of hand deformities in CCA, splint therapy is effective, and the results of surgical treatment including release of the skin contracture and free skin graft were also satisfactory (Ogino et al, 1990). Spinal deformity was a common associated anomaly as in other reports (Shikata et al, 1987;Arroyo et al, 1985).…”

Section: Discussionmentioning

confidence: 89%

“…camptodactyly (Ogino et al, 1989). These hand deformities were often associated with Freeman-Sheldon syndrome, digito-talar dysmorphism.…”

Section: Discussionmentioning

confidence: 99%

“…aplasia of the extensor muscles of the digits were reported (Ogino et al, 1989). CCA was first described by Beak and Hecht (1971) (Bell and Wheller, 1985;Anderson et al, 1984), CCA with eye involvement (Bass et al, 1981;Bard, 1979) and Marfan syndrome with congenital multiple joints contracture (Hibi et al, 1985;Gruber et a!., 1978).…”

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confidence: 99%

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Clinical Features of Congenital Contractural Arachnodactyly

Ogino¹,

Kato²,

Ohshio³

et al. 1993

Congenital Anomalies

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Clinical features of 11 cases of congenital contractural arachnodactyly (CCA) were reported. Eight cases were male and 3 cases were female. Family history was positive in 6 cases of 3 families. As for hand deformity, flexion of the finger, adduction of the thumb and shortening of the palmar skin were observed in all cases, arachnodactyly in ten cases, flexion contracture of the finger joint in 6 cases. Dolichostenomelia and crumpled ear deformity were associated in all cases, spinal deformity in 6 cases, flexion contracture of the lower extremities in 5 cases, micrognathia in 2 cases and high palate, strabismus, pectus excavatum and polydactyly of the fifth toe in one case. Arachnodactyly was not an essential feature of CCA. Existence of multiple camptodactyly with or without arachnodactyly and/or ulnar drift of the fingers, and crumpled ear deformity were important findings for differential diagnosis of CCA. There was no association of cardiovascular anomalies in this series. CCA without ocular and cardiovascular involvement must be a definite clinical entity and should be distinguished from the other forms of marfanoid habitus.

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Section: Discussionmentioning

confidence: 89%

“…camptodactyly (Ogino et al, 1989). These hand deformities were often associated with Freeman-Sheldon syndrome, digito-talar dysmorphism.…”

Section: Discussionmentioning

confidence: 99%

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Clinical Features of Congenital Contractural Arachnodactyly

Ogino¹,

Kato²,

Ohshio³

et al. 1993

Congenital Anomalies

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“…In arthrogryposis multiple congenital, atrophy of muscle is a striking sign and easily differentiated from CMH. In the windblown hand, the thumb in palm deformity is an essential feature, and the thumb is always flexed and adducted (Ogino et al., 1993).…”

Section: Discussionmentioning

confidence: 99%

Morphological and radiological features of congenital muscular hypertrophy of the upper limb: experience from a tertiary institution

Huang,

Zhao,

Sun

et al. 2023

J Hand Surg Eur Vol

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Congenital muscular hypertrophy is a rare overgrowth disorder in the phosphatidylinositol-3-kinase related spectrum. In the past 3 years, ten patients with 11 limbs involved were treated in our centre. The aim of the study was to describe the clinical and radiological deformities of these patients. We documented the characteristic clinical morphological changes, such as hypertrophy, loss of wrist flexion, thumb hyperabduction, finger deviation and skin crease changes in the palm. Radiologically, the mean first metacarpal radial deviation angle of the affected side measured 55° (range 34 to 67) compared to the normal contralateral side 42° (range 32 to 53). The mean intermetacarpal space ratio was 1.2 (range 1.1 to 1.4) and the mean palm width ratio was 1.2 (range 1.1 to 1.3). In this study, we were able to further characterize the radiological and morphological changes of congenital muscular hypertrophy of upper limbs, which would be helpful for establishing the diagnosis and monitor treatment of this rare condition. Level of evidence: IV

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“…They thought that the case reported by So (1992) might be the same condition. In windblown hand, thumb in palm deformity is an essential feature and the thumb is always in flexed and adducted position (Ogino et al . 1993), but in aberrant muscle syndrome, mostly the thumb is in extended and abducted position as shown in our series.…”

Section: Discussionmentioning

confidence: 99%

Aberrant muscle syndrome: Hypertrophy of the hand and arm due to aberrant muscles with or without hypertrophy of the muscles

Ogino

Satake

Takahara

et al. 2010

Congenital Anomalies

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Five patients were reported in our congenital anomaly registry who had six hands in total with muscular hyperplasia, aberrant muscles, ulnar drift of the fingers in the metacarpophalangeal (MP) joints, flexion contractures of the MP joints, and enlargement of the metacarpal spaces. Thirty patients with unilateral involvement of this condition have been reported previously. We reviewed these cases and found that the condition varied in severity and that it was reported using different names. However, this condition seems different from true macrodactyly and multiple camptodactyly, including windblown hand, and seems to be an isolated entity of congenital upper limb anomaly. The authors recommend 'aberrant muscle syndrome' or 'accessory muscle syndrome' as a diagnostic name, because this seems to be the most common pathological finding in this condition.

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Clinical Features and Operative Findings of Congenital Flexion Deformity of Multiple Digits

Cited by 3 publications

References 12 publications

Clinical Features of Congenital Contractural Arachnodactyly

Clinical Features of Congenital Contractural Arachnodactyly

Morphological and radiological features of congenital muscular hypertrophy of the upper limb: experience from a tertiary institution

Aberrant muscle syndrome: Hypertrophy of the hand and arm due to aberrant muscles with or without hypertrophy of the muscles

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