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Radiographic Spectrum of Severity in Madelung's Deformity
SCIENTIFIC ARTICLE
Radiographic Spectrum of Severity in Madelung’s Deformity Dmitry Tuder, MD, Britt Frome, MD, David P. Green, MD
Purpose To establish whether Madelung’s deformity demonstrates a radiographic continuum of severity and whether a forme fruste does exist. Methods Ulnar tilt, lunate subsidence, palmar carpal displacement, and lunate fossa angle were measured in 81 wrist radiographs with obvious or suspected Madelung’s deformity. Statistical analyses based on these measurements were performed to ascertain if there is a deformity continuum. Results Ranges of 15° to 51° (mean, 28°) for ulnar tilt, –7 to ⫹11 mm (mean, – 0.8 mm) for lunate subsidence, 9 to 25 mm (mean, 15.3 mm) for palmar carpal displacement, and 20° to 56° (mean, 33°) for lunate fossa angle were obtained. Significant correlations were observed between all measurements. Conclusions Madelung’s deformity encompasses a spectrum of radiographic abnormality. (J Hand Surg 2008;33A:900– 904. Copyright © 2008 by the American Society for Surgery of the Hand. All rights reserved.) Type of study/level of evidence Diagnostic I. Key words Deformity, Madelung, severity, x-ray.
description in 1878, Madelung’s deformity and its treatment have been described in the literature by multiple authors.1–3 The deformity is a consequence of premature closure of the volar ulnar part of the distal radial physis and can be attributed to genetic, traumatic, or infectious causes.3,4 Twelve radiographic criteria for the diagnosis of Madelung’s deformity were described by Dannenberg et al. in 1939.5 Since then, multiple authors6 –9 have described criteria for the radiographic measurements of the deformity, but no reliability and reproducibility data for these measurements existed until 2005, when Mc-
S
INCE THE ORIGINAL
From the Uniformed Services University, Wilford Hall Medical Center, San Antonio, TX; Legacy Emanuel Hospital, Portland, OR; Department of Orthopaedics, University of Texas Health Science Center at San Antonio, San Antonio, TX; and the Hand Center of San Antonio, San Antonio, TX. Received for publication May 16, 2006; accepted in revised form January 24, 2008. No benefits in any form have been received or will be received related directly or indirectly to the subject of this article. Corresponding author: Dmitry Tuder, MD, Wilford Hall Medical Center, 2200 Bergquist Dr., San Antonio, TX 78236; e-mail: [email protected]. 0363-5023/08/33A06-0016$34.00/0 doi:10.1016/j.jhsa.2008.01.031
900 䉬 © ASSH 䉬 Published by Elsevier, Inc. All rights reserved.
Carroll et al. described 4 reliable and reproducible radiographic measurement techniques for Madelung’s deformity.10 Patients with severe forms of Madelung’s deformity are easily recognized; however, we have encountered patients with seemingly unexplainable cause of wrist pain except for the presence of a slight radiographic abnormality of the ulnar aspect of the articular surface of the distal radius, raising a question of whether there exists a mild (forme fruste) Madelung’s deformity. Retrospective review of a series of x-rays over a 25-year period suggests that the deformity has a spectrum of radiographic severity that could explain our observations. The main purpose of this retrospective radiographic evaluation was to ascertain whether there is a deformity continuum. MATERIALS AND METHODS Patients with the diagnosis of definite or questionable (slight abnormality of the volar aspect of the
SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
FIGURE 1: Ulnar tilt on a posteroanterior x-ray is defined as the complement of the acute angle (angle A) between the longitudinal axis of the ulna and a line tangental to the proximal surfaces of the scaphoid and lunate. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A:1211–1220, with permission of Elsevier.)
lunate fossa of the distal radius) Madelung’s deformity were identified in the database of a single surgeon’s private practice. Fifty-eight patients were included in this evaluation based on the availability of good-quality posteroanterior and lateral wrist plain radiographs. All but 1 patient were female, and 8 patients were skeletally immature. Twenty-three patients had x-rays of both wrists: 21 of the right wrist, and 14 of the left wrist. Therefore, 81 sets of wrist radiographs were available for review. None of the patients in this study had a history of injury or disease in the forearm or wrist. Using the techniques described by McCarroll et al.,10 ulnar tilt, lunate subsidence, palmar carpal displacement, and lunate fossa angle were measured (Figs. 1– 4). Using these values, an analysis including both Pearson (r) and Spearman () correlations was done to determine if the radiographic measures were significantly associated for all patients (whether an increase in one measurement of the deformity is associated with increase in all other measurements). In addition, Cronbach’s ␣ was measured to determine whether all 4 measurements have a good reliability.11 Statistical analyses were performed using statistical
901
FIGURE 2: Lunate subsidence on a posteroanterior x-ray (distance A) is defined as the distance in millimeters between the most proximal point of the lunate and a line perpendicular to the longitudinal axis of the ulna and through its distal articular surface. The measurement is positive if the ulna extends distally to the proximal surface of the lunate. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A: 1211–1220, with permission of Elsevier.)
software (SPSS 10.0, SPSS Inc., Chicago, IL; Stata 9.0, StataCorp LP, College Station, TX). RESULTS The following measurements were obtained in 81 wrists: ulnar tilt range of 15° to 51° (mean, 28°), lunate subsidence range of –7 to ⫹11 mm (mean, –1.0 mm), palmar carpal displacement range of 9 to 25 mm (mean, 15.3 mm), and lunate fossa angle range of 20° to 56° (mean, 33°). For the 81 patients, significant correlations were observed between ulnar tilt and lunate subsidence (r ⫽ 0.50, ⫽ 0.50, p ⬍ .001), between ulnar tilt and palmar carpal displacement (r ⫽ 0.54, ⫽ 0.49, p ⬍ .001), between ulnar tilt and lunate fossa angle (r ⫽ 0.809, ⫽ 0.748, p ⬍ .001), between lunate subsidence and palmar carpal displacement (r ⫽ 0.53, ⫽ 0.46, p ⬍ .001), between lunate subsidence and lunate fossa angle (r ⫽ 0.676, ⫽ 0.692, p ⬍ .001), and between lunate fossa angle and palmar carpal displacement (r ⫽ 0.624, ⫽ .602, p ⬍ .001). Cronbach’s ␣ for all 4 measurements was .827 and did not improve when any of the measurements were removed.
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SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
FIGURE 3: Palmar carpal displacement on a lateral x-ray (distance A) is defined as the distance in millimeters between the longitudinal axis of the ulna and the most palmar point on the surface of the lunate or capitate. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005; 30A:1211–1220, with permission of Elsevier.)
FIGURE 5: Only slight abnormality of the ulnar aspect of the articular surface of the distal radius is noticeable in this radiograph.
FIGURE 6: Radiograph showing increasing lunate fossa angle.
FIGURE 4: Lunate fossa angle on a posteroanterior x-ray is defined as the complement of the acute angle (angle A) between the longitudinal axis of the ulna and a line across the lunate fossa of the radius. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A:1211–1220, with permission of Elsevier.)
DISCUSSION Based on the statistical evaluation of the data, it would appear that Madelung’s deformity encompasses a spectrum of radiographic abnormality (Figs. 5–10). As one of the measurements increased, so did all others. In addition, using all 4 measurements can be considered reliable (Cronbach’s ␣ ⬎ .8). Therefore, just as there
are patients with very severe deformity, there are most likely to be patients with very mild deformity as well. This retrospective radiographic investigation was prompted by a fundamental question: Are these patients with mild deformity more susceptible to wrist pain or injury than are patients with normal wrists? This question was not answered in this study; however, perhaps the recognition of the existence of forme fruste Madelung’s deformity will prompt additional studies. Further clinical studies must be done to determine if this mild degree of deformity is clinically important. In addition, it is not yet known if these most mild forms of the deformity progress; however, most of the patients in this investigation were already skeletally mature, making the possibility of progression doubtful. Additional investigations are needed to further evaluate this. Good-quality, bilateral, neutral wrist, posteroanterior x-rays are important for accurate evaluation and measurements. We recommend the technique described by
JHS 䉬 Vol A, July–August
SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
FIGURE 7: A radiograph showing increasing lunate fossa angle.
FIGURE 8: Radiograph showing that increase in lunate fossa angle and ulnar tilt is becoming more apparent.
Ezaki et al.12 in which the shoulder is abducted 90° and the elbow is flexed 90° as the x-ray beam passes from posterior to anterior. One of the weaknesses of this study is that it examines a relatively small number of subjects. It is not known whether patients who had only unilateral radiographs also had bilateral deformity. The hand dominance of the patients in this study as well as possible associations with Leri-Weill syndrome or other abnormalities is not known. This study shows that “Madelung’s deformity” encompasses a broad spectrum of radiographic abnormality, ranging from very subtle changes in the lunate fossa of the radius to the severe changes easily recognized as Madelung’s deformity. Because this is a continuum, it would be difficult to create a classification system based on arbitrary designations as to degree of radiographic severity.
903
FIGURE 9: Classic Madelung’s deformity with easily recognized abnormalities of lunate fossa angle, ulnar tilt, and lunate subsidence.
FIGURE 10: A radiograph showing classic Madelung’s deformity with easily recognized abnormalities of lunate fossa angle, ulnar tilt, and lunate subsidence.
REFERENCES 1. Madelung O. Die Spontane Subluxation der Hand nach Vorne. Verhandl d deutsch Gesellsch f Chir 1878;7:259 –276 [also Arch f klin Chir 1879;23:395– 412]. 2. Anton JI, Reitz GB, Spiegel MB. Madelung’s deformity. Ann Surg 1938;108:411– 439. 3. Paus B. Madelung’s deformity; its etiology and pathogenesis. Acta Orthop Scand 1951;21:249 –258. 4. Belin V, Cusin V, Viot G, Girlich D, Toutain A, Moncla A, et al. SHOX mutations in dyschondrosteosis (Leri-Weill syndrome). Nat Genet 1998;19:67– 69. 5. Dannenberg M, Anton JI, Spiegel MB. Madelung’s deformity (consideration of its roentgenological diagnostic criteria). Am J Roentgenol Rad Ther 1939;42:671– 676. 6. Ranawat CS, DeFiore J, Straub LR. Madelung’s deformity (an end-result study of surgical treatment). J Bone Joint Surg 1975; 57A:772–775. 7. Salon A, Serra M, Pouliquen JC. Long-term follow-up of surgical correction of Madelung’s deformity with conservation of the distal radioulnar joint in teenagers. J Hand Surg 2000;25B:22–25. 8. dos Reis FB, Katchburian MV, Faloppa F, Albertoni WM, Filho JL.
JHS 䉬 Vol A, July–August
904
SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
Osteotomy of the radius and ulna for the Madelung deformity. J Bone Joint Surg 1998;80B:817– 824. 9. Vickers D, Nielsen G. Madelung deformity (surgical prophylaxis (physiolysis) during the late growth period by resection of the dyschondrosteosis lesion). J Hand Surg 1992;17B:401– 407. 10. McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR.
Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A:1211–1220. 11. Bland JM, Altman DG. Cronbach’s alpha. Brit Med J 1997;314:572– 573. 12. Ezaki M. In: Tachdjian M, ed. Tachdjian’s pediatric orthopaedics. 2nd ed. Philadelphia: W.B. Saunders; 1990:419.
JHS 䉬 Vol A, July–August
Radiographic Spectrum of Severity in Madelung’s Deformity Dmitry Tuder, MD, Britt Frome, MD, David P. Green, MD
Purpose To establish whether Madelung’s deformity demonstrates a radiographic continuum of severity and whether a forme fruste does exist. Methods Ulnar tilt, lunate subsidence, palmar carpal displacement, and lunate fossa angle were measured in 81 wrist radiographs with obvious or suspected Madelung’s deformity. Statistical analyses based on these measurements were performed to ascertain if there is a deformity continuum. Results Ranges of 15° to 51° (mean, 28°) for ulnar tilt, –7 to ⫹11 mm (mean, – 0.8 mm) for lunate subsidence, 9 to 25 mm (mean, 15.3 mm) for palmar carpal displacement, and 20° to 56° (mean, 33°) for lunate fossa angle were obtained. Significant correlations were observed between all measurements. Conclusions Madelung’s deformity encompasses a spectrum of radiographic abnormality. (J Hand Surg 2008;33A:900– 904. Copyright © 2008 by the American Society for Surgery of the Hand. All rights reserved.) Type of study/level of evidence Diagnostic I. Key words Deformity, Madelung, severity, x-ray.
description in 1878, Madelung’s deformity and its treatment have been described in the literature by multiple authors.1–3 The deformity is a consequence of premature closure of the volar ulnar part of the distal radial physis and can be attributed to genetic, traumatic, or infectious causes.3,4 Twelve radiographic criteria for the diagnosis of Madelung’s deformity were described by Dannenberg et al. in 1939.5 Since then, multiple authors6 –9 have described criteria for the radiographic measurements of the deformity, but no reliability and reproducibility data for these measurements existed until 2005, when Mc-
S
INCE THE ORIGINAL
From the Uniformed Services University, Wilford Hall Medical Center, San Antonio, TX; Legacy Emanuel Hospital, Portland, OR; Department of Orthopaedics, University of Texas Health Science Center at San Antonio, San Antonio, TX; and the Hand Center of San Antonio, San Antonio, TX. Received for publication May 16, 2006; accepted in revised form January 24, 2008. No benefits in any form have been received or will be received related directly or indirectly to the subject of this article. Corresponding author: Dmitry Tuder, MD, Wilford Hall Medical Center, 2200 Bergquist Dr., San Antonio, TX 78236; e-mail: [email protected]. 0363-5023/08/33A06-0016$34.00/0 doi:10.1016/j.jhsa.2008.01.031
900 䉬 © ASSH 䉬 Published by Elsevier, Inc. All rights reserved.
Carroll et al. described 4 reliable and reproducible radiographic measurement techniques for Madelung’s deformity.10 Patients with severe forms of Madelung’s deformity are easily recognized; however, we have encountered patients with seemingly unexplainable cause of wrist pain except for the presence of a slight radiographic abnormality of the ulnar aspect of the articular surface of the distal radius, raising a question of whether there exists a mild (forme fruste) Madelung’s deformity. Retrospective review of a series of x-rays over a 25-year period suggests that the deformity has a spectrum of radiographic severity that could explain our observations. The main purpose of this retrospective radiographic evaluation was to ascertain whether there is a deformity continuum. MATERIALS AND METHODS Patients with the diagnosis of definite or questionable (slight abnormality of the volar aspect of the
SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
FIGURE 1: Ulnar tilt on a posteroanterior x-ray is defined as the complement of the acute angle (angle A) between the longitudinal axis of the ulna and a line tangental to the proximal surfaces of the scaphoid and lunate. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A:1211–1220, with permission of Elsevier.)
lunate fossa of the distal radius) Madelung’s deformity were identified in the database of a single surgeon’s private practice. Fifty-eight patients were included in this evaluation based on the availability of good-quality posteroanterior and lateral wrist plain radiographs. All but 1 patient were female, and 8 patients were skeletally immature. Twenty-three patients had x-rays of both wrists: 21 of the right wrist, and 14 of the left wrist. Therefore, 81 sets of wrist radiographs were available for review. None of the patients in this study had a history of injury or disease in the forearm or wrist. Using the techniques described by McCarroll et al.,10 ulnar tilt, lunate subsidence, palmar carpal displacement, and lunate fossa angle were measured (Figs. 1– 4). Using these values, an analysis including both Pearson (r) and Spearman () correlations was done to determine if the radiographic measures were significantly associated for all patients (whether an increase in one measurement of the deformity is associated with increase in all other measurements). In addition, Cronbach’s ␣ was measured to determine whether all 4 measurements have a good reliability.11 Statistical analyses were performed using statistical
901
FIGURE 2: Lunate subsidence on a posteroanterior x-ray (distance A) is defined as the distance in millimeters between the most proximal point of the lunate and a line perpendicular to the longitudinal axis of the ulna and through its distal articular surface. The measurement is positive if the ulna extends distally to the proximal surface of the lunate. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A: 1211–1220, with permission of Elsevier.)
software (SPSS 10.0, SPSS Inc., Chicago, IL; Stata 9.0, StataCorp LP, College Station, TX). RESULTS The following measurements were obtained in 81 wrists: ulnar tilt range of 15° to 51° (mean, 28°), lunate subsidence range of –7 to ⫹11 mm (mean, –1.0 mm), palmar carpal displacement range of 9 to 25 mm (mean, 15.3 mm), and lunate fossa angle range of 20° to 56° (mean, 33°). For the 81 patients, significant correlations were observed between ulnar tilt and lunate subsidence (r ⫽ 0.50, ⫽ 0.50, p ⬍ .001), between ulnar tilt and palmar carpal displacement (r ⫽ 0.54, ⫽ 0.49, p ⬍ .001), between ulnar tilt and lunate fossa angle (r ⫽ 0.809, ⫽ 0.748, p ⬍ .001), between lunate subsidence and palmar carpal displacement (r ⫽ 0.53, ⫽ 0.46, p ⬍ .001), between lunate subsidence and lunate fossa angle (r ⫽ 0.676, ⫽ 0.692, p ⬍ .001), and between lunate fossa angle and palmar carpal displacement (r ⫽ 0.624, ⫽ .602, p ⬍ .001). Cronbach’s ␣ for all 4 measurements was .827 and did not improve when any of the measurements were removed.
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SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
FIGURE 3: Palmar carpal displacement on a lateral x-ray (distance A) is defined as the distance in millimeters between the longitudinal axis of the ulna and the most palmar point on the surface of the lunate or capitate. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005; 30A:1211–1220, with permission of Elsevier.)
FIGURE 5: Only slight abnormality of the ulnar aspect of the articular surface of the distal radius is noticeable in this radiograph.
FIGURE 6: Radiograph showing increasing lunate fossa angle.
FIGURE 4: Lunate fossa angle on a posteroanterior x-ray is defined as the complement of the acute angle (angle A) between the longitudinal axis of the ulna and a line across the lunate fossa of the radius. (Reproduced from McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR. Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A:1211–1220, with permission of Elsevier.)
DISCUSSION Based on the statistical evaluation of the data, it would appear that Madelung’s deformity encompasses a spectrum of radiographic abnormality (Figs. 5–10). As one of the measurements increased, so did all others. In addition, using all 4 measurements can be considered reliable (Cronbach’s ␣ ⬎ .8). Therefore, just as there
are patients with very severe deformity, there are most likely to be patients with very mild deformity as well. This retrospective radiographic investigation was prompted by a fundamental question: Are these patients with mild deformity more susceptible to wrist pain or injury than are patients with normal wrists? This question was not answered in this study; however, perhaps the recognition of the existence of forme fruste Madelung’s deformity will prompt additional studies. Further clinical studies must be done to determine if this mild degree of deformity is clinically important. In addition, it is not yet known if these most mild forms of the deformity progress; however, most of the patients in this investigation were already skeletally mature, making the possibility of progression doubtful. Additional investigations are needed to further evaluate this. Good-quality, bilateral, neutral wrist, posteroanterior x-rays are important for accurate evaluation and measurements. We recommend the technique described by
JHS 䉬 Vol A, July–August
SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
FIGURE 7: A radiograph showing increasing lunate fossa angle.
FIGURE 8: Radiograph showing that increase in lunate fossa angle and ulnar tilt is becoming more apparent.
Ezaki et al.12 in which the shoulder is abducted 90° and the elbow is flexed 90° as the x-ray beam passes from posterior to anterior. One of the weaknesses of this study is that it examines a relatively small number of subjects. It is not known whether patients who had only unilateral radiographs also had bilateral deformity. The hand dominance of the patients in this study as well as possible associations with Leri-Weill syndrome or other abnormalities is not known. This study shows that “Madelung’s deformity” encompasses a broad spectrum of radiographic abnormality, ranging from very subtle changes in the lunate fossa of the radius to the severe changes easily recognized as Madelung’s deformity. Because this is a continuum, it would be difficult to create a classification system based on arbitrary designations as to degree of radiographic severity.
903
FIGURE 9: Classic Madelung’s deformity with easily recognized abnormalities of lunate fossa angle, ulnar tilt, and lunate subsidence.
FIGURE 10: A radiograph showing classic Madelung’s deformity with easily recognized abnormalities of lunate fossa angle, ulnar tilt, and lunate subsidence.
REFERENCES 1. Madelung O. Die Spontane Subluxation der Hand nach Vorne. Verhandl d deutsch Gesellsch f Chir 1878;7:259 –276 [also Arch f klin Chir 1879;23:395– 412]. 2. Anton JI, Reitz GB, Spiegel MB. Madelung’s deformity. Ann Surg 1938;108:411– 439. 3. Paus B. Madelung’s deformity; its etiology and pathogenesis. Acta Orthop Scand 1951;21:249 –258. 4. Belin V, Cusin V, Viot G, Girlich D, Toutain A, Moncla A, et al. SHOX mutations in dyschondrosteosis (Leri-Weill syndrome). Nat Genet 1998;19:67– 69. 5. Dannenberg M, Anton JI, Spiegel MB. Madelung’s deformity (consideration of its roentgenological diagnostic criteria). Am J Roentgenol Rad Ther 1939;42:671– 676. 6. Ranawat CS, DeFiore J, Straub LR. Madelung’s deformity (an end-result study of surgical treatment). J Bone Joint Surg 1975; 57A:772–775. 7. Salon A, Serra M, Pouliquen JC. Long-term follow-up of surgical correction of Madelung’s deformity with conservation of the distal radioulnar joint in teenagers. J Hand Surg 2000;25B:22–25. 8. dos Reis FB, Katchburian MV, Faloppa F, Albertoni WM, Filho JL.
JHS 䉬 Vol A, July–August
904
SPECTRUM OF SEVERITY IN MADELUNG’S DEFORMITY
Osteotomy of the radius and ulna for the Madelung deformity. J Bone Joint Surg 1998;80B:817– 824. 9. Vickers D, Nielsen G. Madelung deformity (surgical prophylaxis (physiolysis) during the late growth period by resection of the dyschondrosteosis lesion). J Hand Surg 1992;17B:401– 407. 10. McCarroll HR, James MA, Newmeyer WL, Molitor F, Manske PR.
Madelung’s deformity: quantitative assessment of x-ray deformity. J Hand Surg 2005;30A:1211–1220. 11. Bland JM, Altman DG. Cronbach’s alpha. Brit Med J 1997;314:572– 573. 12. Ezaki M. In: Tachdjian M, ed. Tachdjian’s pediatric orthopaedics. 2nd ed. Philadelphia: W.B. Saunders; 1990:419.
JHS 䉬 Vol A, July–August