J Korean Assoc Oral Maxillofac Surg.  2017 Jun;43(3):171-177. 10.5125/jkaoms.2017.43.3.171.

Amniotic constriction band: a report of two cases with unique clinical presentations

• Richardson S
• Khandeparker RV
• Pellerin P

• KMID: 2386362
• DOI: http://doi.org/10.5125/jkaoms.2017.43.3.171

Abstract

Amniotic constriction band is a rare clinical entity with varied manifestations that range from a combination of congenital malformations to isolated malformations that are unique to each patient. The etiology of this entity remains unknown. Herein, we highlight two cases of amniotic constriction band that presented to our unit with unique clinical characteristics. To the best of our knowledge, an isolated circumferential band of scarring on the face with ocular involvement, as demonstrated by the first case, and a combination of bilateral complete cleft lip and palate with bilateral microphthalmia, auto-amputation of the right thumb, and a constriction band on the left thumb, as demonstrated by the second case, are extremely rare presentations of amniotic constriction band that were not previously reported in the literature and therefore necessitate a special mention. We discuss potential etiologies for these cases and review the existing literature on this entity.

Keyword

Amniotic constriction band; Circumferential band; Bilateral cleft lip and palate; Auto-amputation; Microphthalmos

MeSH Terms

Cicatrix
Cleft Lip
Constriction*
Humans
Microphthalmos
Palate
Thumb

Figure

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  Fig. 1 Clinical presentation of the circumferential constriction band on the face in Case 1. A. Frontal view showing the constriction band on the face. B. Right lateral view showing the extent of the constriction band on the scalp. C. Left lateral view showing the extent of the constriction band. D. Close-up view to appreciate the ocular involvement.

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  Fig. 2 Three-dimensional computed tomographic view of the facial skeleton in Case 1. A. View demonstrating the smaller dimensions of the right orbit as compared to the left. The right orbit measured 2.9×2.1 cm in greatest dimensions whereas the left orbit measured 3.0×2.6 mm in greatest dimensions. B. View demonstrating a defect in the frontal bone on the left side.

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  Fig. 3 Intraoperative and postoperative photographs of Case 1. A. Intraoperative photographs of bilateral oculoplastic surgery. B. Postoperative photographs at 6 months from surgery.

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  Fig. 4 Clinical presentation of Case 2. A. Frontal view showing bilateral complete cleft lip, premaxillary excess and bilateral microphthalmia. B. Photograph of the right hand showing auto-amputation of the thumb. C. Photograph of the left hand showing constriction band on the left thumb.

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  Fig. 5 Intraoperative and postoperative photographs of bilateral cleft lip repair of Case 2. A. Intraoperative view of repair using Mulliken technique. B. Postoperative view at 6 months.

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  Fig. 6 Intraoperative photograph of cleft palate repair in Case 2.

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  Fig. 7 Intraoperative and postoperative photographs of vomerine osteotomy of Case 2. A. Intraoperative view following osteotomy and fixation. B. Postoperative view.

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