Clinicopathologic Case Report ■ Description clinico-pathologique de cas • Klinisch-pathologische Fallbeschreibung
Opli t halmologica 1992 ;204:210-214
Department of Ophthalmology, Faculty of Medicine, Kyushu University. Maidashi, Fukuoka. Japan
Key Words Goniodysgenesis Arthrogryposis multiplex congenita Infantile glaucoma Histology
Goniodysgenesis of the Eye with Arthrogryposis Multiplex congenita
Abstract
We report an autopsy case of 4-day-old male infant with ar throgryposis multiplex congenita studied by histological meth ods in respect to the ocular tissues. The eye examined was normal in size, and the retina, optic nerve, choroid, sclera and extracocular muscle showed no remarkable histological changes. However, the cornea was thickened, and the iris, trabecular tissue and ciliary body showed maldcvclopmental changes, which were all compatible with goniodysgenesis. The clinical data of this case did not demonstrate any signs of con genital glaucoma, but the present study indicates that an infant with arthrogryposis multiplex congenita has a potential for infantile glaucoma with goniodysgenesis.
Introduction
Arthrogryposis multiplex congenita (AMC) is a syndrome characterized clinically by congenital rigidity and deformation of mul tiple joints. This disorder was first reported by
Ophthalmic pathologists are invited to submitt manuscripts for this section to Prof. Dr. Fritz Stefani. Universitäts-Augenklinik. Mathildenstrasse 9. D-8000 München (FRG). For detailed information see Ophthalmologica 1992:204:103.
Received: August 26, 1991 Accepted: November 12.1991
Otto in 1841 as congenital myodystrophy and has subsequently been frequently described in the literature [1-10], The limitations of joint movement result from various underlying fac tors, such as neuromuscular disorders and ma ternal factors among others. AMC has been described as being associ ated with various kinds of anomalies [1-3]. Among these anomalies, musculoskeletal, ge nitourinary, cardiovascular, gastrointestinal and otorhinolaryngological anomalies have been reported in the literature. From an ophTaiji Sakamoto. MD Department of Ophthalmology Faculty of Medicine. Kyushu University 60 3-1-1 Maidashi. Higashi-ku Fukuoka 812 (Japan)
© 1992 S. Karger AG. Basel 0030-3755/92/20440210$2.75A)
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
Taiji Sakamoto Akihiko Tawara Hajime Inom ata
a
Fig. 1. Photographs of the pa tient at autopsy, a Flexion contrac tions of both feet and hands are no ticed. b Low-set ears and webbed neck are seen.
Case Report A 2-day-old male infant was hospitalized with se vere postural anomalies of the limbs and sucking prob lems. He was delivered by cesarean section and was the product of a 37-week uneventful gestation. At birth, which was in a breech presentation, the infant weighed 2.700 g. The Apgar score was 10 at 1 min. Multiple skeletal distortions were observed at delivery. Flexion contractures of both feet and hands, cleft palate, lowset ears, micrognathia and short neck were markedly present. At 3 days after birth, the abdomen became
distended, and an intestinal obstruction with incarcer ated right inguinal hernia was found. Although the hernia was repaired. he developed acute peritonitis and died of respiratory disorder 4 days after birth. The laboratory data at 3 days disclosed the following: WBC 12,400/mnr'; RBC 4,790,000/dl: Hb 17.2 mg/dl: Hct 52.5%, and platelets 317,000/dl. His mother was 22 years old, and neither of the parents had any family histories of congenital anoma lies including AMC or neuromuscular disease. This was her first gestation. General Autopsy Findings The autopsy was performed 10 h after death. The patient was 51 cm in length and weighed 2,600 g. The patient had both clubhands and clubfeet, and in addi tion flexion of knees, abduction and flexion at both hips, cleft palate, low-set ears, micrognathia and webbed neck were noted (fig. la,b). The lungs, liver, spleen and kidneys showed a normal maturation for a baby of 37 weeks gestational age, and no serious ab normalities regarding cryptorchidism or any atrial sep tal defect of the heart were present. The brain weighed 320g and showed a normal maturation in size and sur face configuration. The cecum was herniated into the inguinal canal and a part of its wall was perforated associated with depositions of necrotized mass and ab scesses.
211
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thalmological point of view, Moebius syn drome, hypertelorism, microphthalmia, con genital glaucoma, congenital cataract, lateral rectus muscle palsy, lid anomalies and optic atrophy have all been observed clinically [1,4, 5, 8]. However, to our knowledge, goniodysgenesis associated with AMC and its histopathological features have not yet been re ported. We herein present the unusual histo logical findings of an eye with arthrogryposis from an autopsy case.
Fig. 2. Photomicrograph of the anterior portions of the left eye. The cornea is thick while the corneal endothelium is partly denuded. The lens is located an teriorly to the cornea. Hematoxylin and eosin. x 4.
Fig. 3. Photomicrograph of the left eye. The hypoplastic iris adheres to the lens epithelium at the pupullary margin. No blood vessels are present at the anterior surface of the lens. Hematoxylin and eosin. x8.
Ocular Findings Macroscopically, the cornea of the right eye w;as clear and showed no serious changes, but a central lesion of the left cornea was clouded and edematous. The diameter of both corneas were equally 9 mm. The left eye was enucleated, fixed in 10% paraformalde
212
Sakamoto/Tawara/I nomata
Goniodysgenesis of Arthrogryposis Multiplex congenita
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
Fig. 4. Photomicrograph of the left eye at the anterior chamber angle. Spindle-shaped mesenchymal cells are found at the angle. Schlemm's canal is not clear but a small vessel is noticed (arrow). The cliliary body and trabeculum are hypoplastic. The trabecular beams are thinner and sparser than those of an infant of the same gestational age. Hematoxylin and eosin. x 20.
hyde and examined microscopically. The left eye had a diam eterof!5 mm and weighed 2,600 mg (normal at 37 weeks: 15 mm, 2,800 mg). Microscopically, the center of the posterior cornea was edematously thickened in part and the endotheli um was partially denuded (fig. 2). The anterior iris stroma was hypoplastic. The lens w'as displaced to the anterior chamber, and the anterior surface of the lens adhered to the posterior iris pigment epithelium con tinuously. but no blood vessels were seen at the anterior or posterior surface of the lens or vitreous (fig. 3). The axial portion of the lens was cone-shaped and the ante rior lenticular capsule disappeared there (fig. 2,4). The anterior chamber angle recess was composed of a loose network of mesenchymal cells (fig. 4). No trabeculae of the uveal or corneoscleral m eslwork were present in the region (fig. 4). Although there was no Schlemm's canal identified, a vessel with a small lumen wasobserved at the apex of the angle (fig. 4, arrow). The wall of the vessel was covered by a layer of cells, but no giant vacuole was observed on it. The scleral spur w'as ob scure. The corneal endothelium was continuous to the mesenchymal cells of the apex, but Schwalbe's line w'as unclear. The retina, choroid, optic nerve and extraoc ular muscle showed a normal maturation and no histo logical abnormality.
AMC is a nonprogressive congenital syn drome complex characterized by poorly de veloped and contracted muscle and joints [1-3]. This syndrome is multifactorial in etiol ogy and has been reported to be associated with a variety of other congenital anomalies [1-9]. From an opthalmological viewpoint. AMC has been described as being associated with microphthalmia, cataracts, glaucoma and cor neal opacities[l, 4.9]. Indeed, such conditions might be common problems for obtaining good vision in AMC children. However, those reports were documented mainly from a clin ical point of view, so that the pathological findings or pathogenesis of the disease have yet to be elucidated. This case was diagnosed as having AMC because of its deformity of multiple joints as well as its association with multiple anomalies which are all common in AMC. such as low-set ears, cleft palate, short neck, micrognathia, inguinal hernia, cryptor chidism and atrial septal defect. On examination of the eyeball, thickness of the cornea and maldevelopment of the tis sues of the anterior segment including the tra becular meshwork, iris and ciliary body were demonstrated. Although the localized coneshaped protrusion and the denudation of the anterior lenticular capsule of the axial portion were similar to those of the anterior lentiglobus, the possibility still remained that those were the results of an artifact of the fixation procedure. Infantile glaucomas have also been path ologically documented by some authors [1014]. According to previous studies, the trabec ular tissue has a well-developed Schlemm’s canal with giant vacuoles in fetal eyes of 37 gestational weeks. The posterior edge of the canal lies at the level of the apex of the ante rior chamber. The uveal and corneoscleral tra
becular meshworks are well organized at this stage. In our case, a vessel, the nature of which was not clearly identified, was observed in the region of the anterior chamber angle. The lumen of the vessel was small and located at the apex of the angle. If the vessel is not Schlemm’s canal, the absence of the canal in dicates that the anterior chamber is prema ture. Even if the vessel were Schlemm's canal, the tissue of the anterior chamber angle is pre mature because of its size. The absence of the uveal and corneoscleral trabeculae also sup ports that the tissue is underdeveloped as a 37-gestational-week chamber angle. The cili ary body muscles and the trabecular mesh work were also not well developed as com pared to those of normal infants at the same gestational age. The findings were also similar to those in patients with fetal anterior cham ber angle and infantile glaucoma. Unfortu nately. this case had not been examined ophthalmologically in detail before his death so it is still unclear whether he had glaucoma or not, but the histological findings of this case were so similar to those of infantile glaucoma that it might be reasonable to say, at least, that this case would have had a strong potential for glaucoma. There are many pathological conditions which lead to glaucomas [10]. Zeiter and Boniuk [8] reported one case of primary openangle glaucoma with AMC. but none of the previous authors who reported glaucomas with AMC up to now referred to the types or pathogenesis of glaucoma with AMC [1.4, 5, 8]. From the present study, we can assert the clear potential of some AMC patients to ei ther have or develop infantile glaucoma with goniodysgenesis. AMC is a multifactorial dis ease; it is believed to be far more neurogenic than myogenic diseases and is associated with a variety of neurogenic disorders [4, 6, 7, 9], The prevalence of goniodysgenesis in this dis ease is understandable because the ciliary
213
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
Discussion
body, trabecular meshwork and iris are all de rived from the mesenchymal cells of the neu ral crest [15]. Therefore, glaucoma might be associated more often than would normally be expected. The principal goal of treatment in AMC is to achieve the maximum function possible for
each organ. For the purpose of obtaining a good vision for AMC children, we must take care not only to perform routine slitlamp or funduscopic examinations but gonioscopic ex amination as well in order to not miss any pre sent glaucoma or future glaucoma and thus develop better strategies for treating them.
1 Swinyard CA, Mayer V: Multiple congenital contractures; public health considerations of arthrogry posis multiplex congenita. JAMA 1963;183:23-27. 2 Brown I.M. Sharrard WJW: The pathophysiology of arthrogryposis multiplex congenita neurologica. J Bone Joint Surg 1980;62B:291. 3 Thompson GH. Bilenker RM: Comprehensive management of ar throgryposis multiplex congenita. Clin Orthop Relat Res 1985:194:614. 4 Banker BO: Arthrogryposis multi plex congenita: Spectrum of path ologic changes. Hum Pathol 1986; 17:656-672.
5 Paez JH. Tuulonen A, Yarom R. Arad I. Zelikovitch A. BenEzra D: Ocular findings in arthrogryposis multiplex congenita. J Pediatr Oph thalmol Strabismus 1982:19:75— 79. 6 Rosenmann A. Arad I: Arthrogry posis multiplex congenita: Neuro logic type with autosomal recessive inheritance. J Med Genet 1974:11: 91-93.
214
7 Banker BQ: Neuropathologie as pects of arthrogryposis multiplex congenita. Clin Orthop Relat Res 1985:194:30-43. 8 Zeiter JH. Boniuk M: Ophthalmo logic findings associated with ar throgryposis multiplex congenita: Case report and review of the litera ture. J Pediatr Ophthalmol Strabis mus 1989:26:204-208. 9 Fowler M: A case of arthrogryposis multiplex congenita with lesions in the nervous system. Arch Dis Child 1959:34:505-510. 10 Schields MB. Ritch R. Krupin T: Classification and mechanism of the glaucomas; in Ritch R. Schields MB. Krupin T (eds): The Glauco mas. St. Louis. Mosby. 1989. vol2. pp 751-755. 11 Anderson DR: The development of the trabecular meshwork and its ab normality in primary infantile glau coma. Trans Am Opthalmol Soc 1981:79:458-485.
12 Jerndal T: Goniodysgenesis and congenital glaucoma: in Jerndal T. Hansson H. Bill A (eds): Goniodys genesis: A new prospective on glau coma. Copenhagen, Scriptor. 1978, pp 67-90. 13 Smelser GK. Ozanics V: The devel opment of the trabecular meshwork in primates. Am J Opthalmol 1971; 71:366-385. 14 Tawara A. Inomata H: Develop mental immaturity of the trabecular meshwork in congenital glaucoma. Am J Ophthalmol 1981:92:508-525. 15 Noden DM: Periocular mesen chyme: Neural crest and mesenchy mal interactions: in Jacobiec FA (ed): in Ocular Anatomy. Embryol ogy and Teratology. Philadelphia. Harper & Row, 1982. pp 97-119.
Sakamoto/Taw'ara/Inomata
Goniodysgenesis of Arthrogryposis Multiplex congenita
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
References
Opli t halmologica 1992 ;204:210-214
Department of Ophthalmology, Faculty of Medicine, Kyushu University. Maidashi, Fukuoka. Japan
Key Words Goniodysgenesis Arthrogryposis multiplex congenita Infantile glaucoma Histology
Goniodysgenesis of the Eye with Arthrogryposis Multiplex congenita
Abstract
We report an autopsy case of 4-day-old male infant with ar throgryposis multiplex congenita studied by histological meth ods in respect to the ocular tissues. The eye examined was normal in size, and the retina, optic nerve, choroid, sclera and extracocular muscle showed no remarkable histological changes. However, the cornea was thickened, and the iris, trabecular tissue and ciliary body showed maldcvclopmental changes, which were all compatible with goniodysgenesis. The clinical data of this case did not demonstrate any signs of con genital glaucoma, but the present study indicates that an infant with arthrogryposis multiplex congenita has a potential for infantile glaucoma with goniodysgenesis.
Introduction
Arthrogryposis multiplex congenita (AMC) is a syndrome characterized clinically by congenital rigidity and deformation of mul tiple joints. This disorder was first reported by
Ophthalmic pathologists are invited to submitt manuscripts for this section to Prof. Dr. Fritz Stefani. Universitäts-Augenklinik. Mathildenstrasse 9. D-8000 München (FRG). For detailed information see Ophthalmologica 1992:204:103.
Received: August 26, 1991 Accepted: November 12.1991
Otto in 1841 as congenital myodystrophy and has subsequently been frequently described in the literature [1-10], The limitations of joint movement result from various underlying fac tors, such as neuromuscular disorders and ma ternal factors among others. AMC has been described as being associ ated with various kinds of anomalies [1-3]. Among these anomalies, musculoskeletal, ge nitourinary, cardiovascular, gastrointestinal and otorhinolaryngological anomalies have been reported in the literature. From an ophTaiji Sakamoto. MD Department of Ophthalmology Faculty of Medicine. Kyushu University 60 3-1-1 Maidashi. Higashi-ku Fukuoka 812 (Japan)
© 1992 S. Karger AG. Basel 0030-3755/92/20440210$2.75A)
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
Taiji Sakamoto Akihiko Tawara Hajime Inom ata
a
Fig. 1. Photographs of the pa tient at autopsy, a Flexion contrac tions of both feet and hands are no ticed. b Low-set ears and webbed neck are seen.
Case Report A 2-day-old male infant was hospitalized with se vere postural anomalies of the limbs and sucking prob lems. He was delivered by cesarean section and was the product of a 37-week uneventful gestation. At birth, which was in a breech presentation, the infant weighed 2.700 g. The Apgar score was 10 at 1 min. Multiple skeletal distortions were observed at delivery. Flexion contractures of both feet and hands, cleft palate, lowset ears, micrognathia and short neck were markedly present. At 3 days after birth, the abdomen became
distended, and an intestinal obstruction with incarcer ated right inguinal hernia was found. Although the hernia was repaired. he developed acute peritonitis and died of respiratory disorder 4 days after birth. The laboratory data at 3 days disclosed the following: WBC 12,400/mnr'; RBC 4,790,000/dl: Hb 17.2 mg/dl: Hct 52.5%, and platelets 317,000/dl. His mother was 22 years old, and neither of the parents had any family histories of congenital anoma lies including AMC or neuromuscular disease. This was her first gestation. General Autopsy Findings The autopsy was performed 10 h after death. The patient was 51 cm in length and weighed 2,600 g. The patient had both clubhands and clubfeet, and in addi tion flexion of knees, abduction and flexion at both hips, cleft palate, low-set ears, micrognathia and webbed neck were noted (fig. la,b). The lungs, liver, spleen and kidneys showed a normal maturation for a baby of 37 weeks gestational age, and no serious ab normalities regarding cryptorchidism or any atrial sep tal defect of the heart were present. The brain weighed 320g and showed a normal maturation in size and sur face configuration. The cecum was herniated into the inguinal canal and a part of its wall was perforated associated with depositions of necrotized mass and ab scesses.
211
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thalmological point of view, Moebius syn drome, hypertelorism, microphthalmia, con genital glaucoma, congenital cataract, lateral rectus muscle palsy, lid anomalies and optic atrophy have all been observed clinically [1,4, 5, 8]. However, to our knowledge, goniodysgenesis associated with AMC and its histopathological features have not yet been re ported. We herein present the unusual histo logical findings of an eye with arthrogryposis from an autopsy case.
Fig. 2. Photomicrograph of the anterior portions of the left eye. The cornea is thick while the corneal endothelium is partly denuded. The lens is located an teriorly to the cornea. Hematoxylin and eosin. x 4.
Fig. 3. Photomicrograph of the left eye. The hypoplastic iris adheres to the lens epithelium at the pupullary margin. No blood vessels are present at the anterior surface of the lens. Hematoxylin and eosin. x8.
Ocular Findings Macroscopically, the cornea of the right eye w;as clear and showed no serious changes, but a central lesion of the left cornea was clouded and edematous. The diameter of both corneas were equally 9 mm. The left eye was enucleated, fixed in 10% paraformalde
212
Sakamoto/Tawara/I nomata
Goniodysgenesis of Arthrogryposis Multiplex congenita
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
Fig. 4. Photomicrograph of the left eye at the anterior chamber angle. Spindle-shaped mesenchymal cells are found at the angle. Schlemm's canal is not clear but a small vessel is noticed (arrow). The cliliary body and trabeculum are hypoplastic. The trabecular beams are thinner and sparser than those of an infant of the same gestational age. Hematoxylin and eosin. x 20.
hyde and examined microscopically. The left eye had a diam eterof!5 mm and weighed 2,600 mg (normal at 37 weeks: 15 mm, 2,800 mg). Microscopically, the center of the posterior cornea was edematously thickened in part and the endotheli um was partially denuded (fig. 2). The anterior iris stroma was hypoplastic. The lens w'as displaced to the anterior chamber, and the anterior surface of the lens adhered to the posterior iris pigment epithelium con tinuously. but no blood vessels were seen at the anterior or posterior surface of the lens or vitreous (fig. 3). The axial portion of the lens was cone-shaped and the ante rior lenticular capsule disappeared there (fig. 2,4). The anterior chamber angle recess was composed of a loose network of mesenchymal cells (fig. 4). No trabeculae of the uveal or corneoscleral m eslwork were present in the region (fig. 4). Although there was no Schlemm's canal identified, a vessel with a small lumen wasobserved at the apex of the angle (fig. 4, arrow). The wall of the vessel was covered by a layer of cells, but no giant vacuole was observed on it. The scleral spur w'as ob scure. The corneal endothelium was continuous to the mesenchymal cells of the apex, but Schwalbe's line w'as unclear. The retina, choroid, optic nerve and extraoc ular muscle showed a normal maturation and no histo logical abnormality.
AMC is a nonprogressive congenital syn drome complex characterized by poorly de veloped and contracted muscle and joints [1-3]. This syndrome is multifactorial in etiol ogy and has been reported to be associated with a variety of other congenital anomalies [1-9]. From an opthalmological viewpoint. AMC has been described as being associated with microphthalmia, cataracts, glaucoma and cor neal opacities[l, 4.9]. Indeed, such conditions might be common problems for obtaining good vision in AMC children. However, those reports were documented mainly from a clin ical point of view, so that the pathological findings or pathogenesis of the disease have yet to be elucidated. This case was diagnosed as having AMC because of its deformity of multiple joints as well as its association with multiple anomalies which are all common in AMC. such as low-set ears, cleft palate, short neck, micrognathia, inguinal hernia, cryptor chidism and atrial septal defect. On examination of the eyeball, thickness of the cornea and maldevelopment of the tis sues of the anterior segment including the tra becular meshwork, iris and ciliary body were demonstrated. Although the localized coneshaped protrusion and the denudation of the anterior lenticular capsule of the axial portion were similar to those of the anterior lentiglobus, the possibility still remained that those were the results of an artifact of the fixation procedure. Infantile glaucomas have also been path ologically documented by some authors [1014]. According to previous studies, the trabec ular tissue has a well-developed Schlemm’s canal with giant vacuoles in fetal eyes of 37 gestational weeks. The posterior edge of the canal lies at the level of the apex of the ante rior chamber. The uveal and corneoscleral tra
becular meshworks are well organized at this stage. In our case, a vessel, the nature of which was not clearly identified, was observed in the region of the anterior chamber angle. The lumen of the vessel was small and located at the apex of the angle. If the vessel is not Schlemm’s canal, the absence of the canal in dicates that the anterior chamber is prema ture. Even if the vessel were Schlemm's canal, the tissue of the anterior chamber angle is pre mature because of its size. The absence of the uveal and corneoscleral trabeculae also sup ports that the tissue is underdeveloped as a 37-gestational-week chamber angle. The cili ary body muscles and the trabecular mesh work were also not well developed as com pared to those of normal infants at the same gestational age. The findings were also similar to those in patients with fetal anterior cham ber angle and infantile glaucoma. Unfortu nately. this case had not been examined ophthalmologically in detail before his death so it is still unclear whether he had glaucoma or not, but the histological findings of this case were so similar to those of infantile glaucoma that it might be reasonable to say, at least, that this case would have had a strong potential for glaucoma. There are many pathological conditions which lead to glaucomas [10]. Zeiter and Boniuk [8] reported one case of primary openangle glaucoma with AMC. but none of the previous authors who reported glaucomas with AMC up to now referred to the types or pathogenesis of glaucoma with AMC [1.4, 5, 8]. From the present study, we can assert the clear potential of some AMC patients to ei ther have or develop infantile glaucoma with goniodysgenesis. AMC is a multifactorial dis ease; it is believed to be far more neurogenic than myogenic diseases and is associated with a variety of neurogenic disorders [4, 6, 7, 9], The prevalence of goniodysgenesis in this dis ease is understandable because the ciliary
213
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
Discussion
body, trabecular meshwork and iris are all de rived from the mesenchymal cells of the neu ral crest [15]. Therefore, glaucoma might be associated more often than would normally be expected. The principal goal of treatment in AMC is to achieve the maximum function possible for
each organ. For the purpose of obtaining a good vision for AMC children, we must take care not only to perform routine slitlamp or funduscopic examinations but gonioscopic ex amination as well in order to not miss any pre sent glaucoma or future glaucoma and thus develop better strategies for treating them.
1 Swinyard CA, Mayer V: Multiple congenital contractures; public health considerations of arthrogry posis multiplex congenita. JAMA 1963;183:23-27. 2 Brown I.M. Sharrard WJW: The pathophysiology of arthrogryposis multiplex congenita neurologica. J Bone Joint Surg 1980;62B:291. 3 Thompson GH. Bilenker RM: Comprehensive management of ar throgryposis multiplex congenita. Clin Orthop Relat Res 1985:194:614. 4 Banker BO: Arthrogryposis multi plex congenita: Spectrum of path ologic changes. Hum Pathol 1986; 17:656-672.
5 Paez JH. Tuulonen A, Yarom R. Arad I. Zelikovitch A. BenEzra D: Ocular findings in arthrogryposis multiplex congenita. J Pediatr Oph thalmol Strabismus 1982:19:75— 79. 6 Rosenmann A. Arad I: Arthrogry posis multiplex congenita: Neuro logic type with autosomal recessive inheritance. J Med Genet 1974:11: 91-93.
214
7 Banker BQ: Neuropathologie as pects of arthrogryposis multiplex congenita. Clin Orthop Relat Res 1985:194:30-43. 8 Zeiter JH. Boniuk M: Ophthalmo logic findings associated with ar throgryposis multiplex congenita: Case report and review of the litera ture. J Pediatr Ophthalmol Strabis mus 1989:26:204-208. 9 Fowler M: A case of arthrogryposis multiplex congenita with lesions in the nervous system. Arch Dis Child 1959:34:505-510. 10 Schields MB. Ritch R. Krupin T: Classification and mechanism of the glaucomas; in Ritch R. Schields MB. Krupin T (eds): The Glauco mas. St. Louis. Mosby. 1989. vol2. pp 751-755. 11 Anderson DR: The development of the trabecular meshwork and its ab normality in primary infantile glau coma. Trans Am Opthalmol Soc 1981:79:458-485.
12 Jerndal T: Goniodysgenesis and congenital glaucoma: in Jerndal T. Hansson H. Bill A (eds): Goniodys genesis: A new prospective on glau coma. Copenhagen, Scriptor. 1978, pp 67-90. 13 Smelser GK. Ozanics V: The devel opment of the trabecular meshwork in primates. Am J Opthalmol 1971; 71:366-385. 14 Tawara A. Inomata H: Develop mental immaturity of the trabecular meshwork in congenital glaucoma. Am J Ophthalmol 1981:92:508-525. 15 Noden DM: Periocular mesen chyme: Neural crest and mesenchy mal interactions: in Jacobiec FA (ed): in Ocular Anatomy. Embryol ogy and Teratology. Philadelphia. Harper & Row, 1982. pp 97-119.
Sakamoto/Taw'ara/Inomata
Goniodysgenesis of Arthrogryposis Multiplex congenita
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 3/3/2018 1:19:53 PM
References
