1976, British Journal of Radiology, 49, 94-96 Case reports
(3) A plexiform type where the sinus is composed of a multitude of small veins with rich anastomoses. The lumen of the sinus is traversed by trabeculae giving it the appearance of spongy tissue. Although the right cavernous sinus in this patient appears to consist of multiple channels, the calcification demonstrated within it must be considered as being pathological. Haemangiomata and varices in the orbit are well described (Viallemonte et al., 1959). In some patients a venous malformation may extend outside the orbit into the middle fossa (Hanafee, Sing and Dayton, 1968), through an enlarged sphenoidal fissure (Brauston and Norton, 1963), onto the scalp, frontal bone or hard palate (Lloyd, Wright and Norman, 1971). Lloyd (1974) also describes two patients in whom a haemangioma within the muscle cone was associated with abnormal veins remote from the site of the haemangioma. In both patients these veins lay on the lateral orbital wall. The patient being described showed no evidence of a venous abnormality within the orbit and there was no evidence of an anomaly elsewhere in the skull or face. The condition at the present time is stable and surgery is not contemplated in the immediate future.
CONCLUSIONS
A hitherto undescribed entity of a venous malformation of the right cavernous sinus is described in a 14-year-old boy with proptosis and evidence of involvement of the right sympathetic and third and sixth cranial nerves. The lesion is confined to the cavernous sinus and there is no evidence of a concomitant intra- or extra-orbital venous malformation or of an intra-cranial arterio-venous anomaly. ACKNOWLEDGMENT
We are indebted to Dr. G. Lloyd for his valuable opinion on the radiological changes in this case.
REFERENCES BRAUSTON, B. B., and NORTON, E. N. D., 1963. Intermittent
exophthalmos. American Journal of Ophthalmology, 55, 701-708. HANAFEE, W. N., SING, P. C , and DAYTON, G. O., 1968.
Orbital venography. American Journal of Roentgenology, 104, 29-35. LLOYD, G. A. S., WRIGHT, J. E., and MORGAN, G., 1971.
Venous malformation in the orbit. British Journal of Ophthalmology, 55, 505-516. LLOYD, G. A. S., 1974. Pathological veins in the orbit. British Journal ofRadiology, 47, 570-578. VIALLEFONT, H., PALEIRAC, R., and BOUDET, C , 1959.
The radiological examination of the cavernous sinuses. Bulletin de la Societe Franc Opthalmology 72, 200-219.
Renal cortical necrosis and papillary necrosis in an infant By M. R. Funston, M.B.B.Ch., D.M.R.D., B. J. Cremin, F.R.A.C.R., F.R.C.R., and I. J. K. Tidbury M.B.Ch.B. Department of Radiology, Groote Schuur Hospital, Observatory 7925, Cape Town, So uth Africa {Submitted March, 1975 and in revised form May, 1975)
Bilateral renal cortical necrosis (BRCN) and renal papillary necrosis have been reported in infancy as a complication of gastro-enteritis, perinatal asphyxia, utero-placental haemorrhage, septicaemia, exsanguination, kernicterus, eclampsia, renal vein thrombosis, fetomaternal transfusion and anoxic shock (Mauer and Nogrady, 1969; Leonidas, Berdon and Gribetz, 1971). We wish to report a case of combined BRCN and papillary necrosis, with some unusual radiological findings. Case history A female infant aged three months presented with a fourday history of severe gastro-enteritis followed by convulsions. She was oliguric with metabolic acidosis. The urine contained protein and red cells. The blood urea was 184 and
the serum creatinine 2-5 mg/100 ml. The patient had a spontaneous diuresis without requiring dialysis. In the diuretic phase fluid, electrolyte and acid-base balance was corrected with difficulty. Four weeks after the onset of the illness, an excretory urogram was performed. At the time of the examination the infant was well hydrated, polyuric, and 20 ml. of sodium iothalamate 54 per cent W/V (Conray 325) was injected intravenously. No calcification was present on the control film of the abdomen. Both kidneys were enlarged, measuring 7-5 cm in length. Prompt opacification of the calyces was seen, and all the calyces were noted to be dilated. The ureters were of normal calibre and there was no ureteric or bladder outlet obstruction. A striking feature was dense opacification of the renal cortex and the columns of Bertin, which occurred five minutes after injection of the contrast. The medullary zones were distinctly less opacified. This difference in opacification of the cortex and medulla persisted for 30 minutes. After one hour there was a steady increase in overall kidney density, with equal opacification of the cortex and medulla, which persisted for four days (Fig. 1).
94
FIG. 1. Excretory urogram (four-day film) showing enlargement of the kidneys and a prolonged dense nephrogram.
At the time of this excretory urogram the patient was in the diuretic phase of acute renal failure with a blood urea of 205 mg/100 ml., and there was no reduction in urine output during or after urography. A retrograde uretero-pyelogram showed that the ureters were of normal calibre but bilateral caliectasis was again demonstrated. The absence of ureteric or bladder outlet obstruction was confirmed. Nine weeks after the onset of the illness, bilateral "tramline" cortical calcification, with calcification of the columns of Bertin, was observed on a plain radiograph of the abdomen (Fig. 2). This calcification was only demonstrated with the patient prone and using a low kilovoltage technique. The calcification had completely disappeared 12 weeks later, and a repeat excretory urogram at this stage revealed the typical calyceal changes of papillary necrosis. There was a marked reduction in renal size when compared with the initial urogram. At this time the patient's renal function had returned to normal.
FIG. 2. LogEtronic print of a radiograph of the upper abdomen showing cortical calcification of the right kidney.
The dilatation of the calyces without evidence of obstruction observed in the initial urogram in our patient was considered to be due to swelling of the papillae as a result of papillary necrosis. Changes observed in the subsequent urogram were typical of papillary necrosis, with evidence of sloughed papillae and a reduction in size. Chrispin et al. (1970) and Chrispin (1972) reported heavy opacification by contrast medium of the medullary zones in medullary necrosis. A uniformly dense nephrogram soon after the injection of contrast medium has been reported in patients with acute tubular necrosis by Fry and Cattell (1972). This phenomenon is thought to be the result of leakage of contrast medium into the interstitial space of the kidney. In our patient, however, the heavy opacification occurred in the cortex and the septal columns of Bertin, while the medullary zones appeared relatively radio-translucent. This appearance has not been previously reported. One can only postulate that the degree of tubular damage in the cortex was greater than in the medulla, allowing a greater amount of leakage to occur into the interstitial space of the cortex. The prolonged dense nephrogram, despite prompt calyceal filling, observed in our patient is thought to be the result of the same process, viz. leakage into the interstitial space. Berdon et al. (1969) have reported similar findings. These authors speculated
DISCUSSION
There are numerous reports in the literature of bilateral renal cortical necrosis in infancy (Leonidas et al, 1971; Eskeland and Skogrand, 1959; Mauer and Nogrady, 1969), but the radiographic finding of cortical calcification following this disease in infancy has rarely been observed. Leonidas et al. (1971) and Whelan, Ling and Davis (1967) reported cases of bilateral renal cortical necrosis in infancy followed by cortical calcification. The cortical calcification observed in our patient was not visible on a radiograph exposed with the usual radiographic factors employed for a plain film of the abdomen in an infant. It was only demonstrated using a low kilovoltage technique, with the patient in the prone position, and was not observed in the radiograph taken three months later. We speculate, therefore, that there may be two reasons why this calcification is not more frequently reported in infancy: optimum radiographic factors are not employed, and the calcification may often be temporary. 95
VOL.
49, No. 577 Case reports
that Tamm-Horsfall protein has been precipitated by contrast medium, resulting in tubular blockage and producing a uniformly prolonged tubular nephrogram. However, all their patients were oliguric at the time of excretory urography, whereas our patient was well-hydrated and polyuric. It is therefore unlikely that protein-precipitation was the cause for the prolonged nephrogram in our patient.
CHRISPIN, A. R., 1972. Medullary necrosis in infancy. British Medical Bulletin, 28, 233-236.
ACKNOWLEDGMENTS
LEONIDAS, J. C , BERDON, W. E., and GRIBETZ, D., 1971.
CHRISPIN, A. R., HULL, D., LILLIE, J. G., and RIDSON,
R. A., 1970. Renal tubular necrosis and papillary necrosis after gastro-enteritis in infants. British Medical journal, 1, 410-412. ESKELAND, G., and SKOGRAND, A., 1959. Bilateral cortical
necrosis of the kidneys in infancy. Acta Paediatrica, 48, 278-286. FRY, I. K., and CATTELL, W. R., 1972. The nephrographic
pattern during excretion urography. British Medical Bulletin, 28, 227-232. Bilateral renal cortical necrosis in the newborn infant: Roentgenographic diagnosis. The Journal of Paediatrics, 79, 623-627.
This case was examined during a visit of Dr. W. Berdon of Columbia University and Dr. G. Friedland of the Bowman Grey School of Medicine. They were both most helpful in corroborating our diagnosis, and we are grateful to Dr. Friedland for his suggestion of taking the prone low kilovoltage film, and his help in acquiring the LogEtronic reproduction of Fig. 2.
MCALLISTER, W. H., and NEDELMAN, S. H., 1961.
The
Roentgen manifestations of bilateral renal cortical necrosis. American Journal of Roentgenology, 86,129. MAUER, S. M., and NOGRADY, M. B., 1969. Renal papillary
and cortical necrosis in a newborn infant: Report of a survivor with roentgenolic documentation. The Journal of Paediatrics, 14, 750-754.
REFERENCES BERDON, W. E., SCHWARTZ, R. H., BECKER, J., and BAKER,
D. H., 1969. Tamm-Horsfall proteinuria. Its relationship to prolonged nephrogram in infants and children and to renal failure following intravenous urography in adults with multiple myeloma. Radiology, 92, 714-722.
WHELAN, J. G., LING, J. T., and
DAVIS, L. A.,
1967.
Antemortem Roentgen manifestations of bilateral renal cortical necrosis. Radiology, 89, 682-689.
Book reviews Radionuclide Scanning in Cyanotic Heart Disease. By Gary F. Gates, M.D., pp. xi +100,1974. (Charles C. Thomas, Illinois, USA), $15.00. The earnest reader who sees the title of this book might be misled into thinking that he was about to possess a monograph covering a subject: the use of radionuclides as a noninvasive way of investigating cyanotic congenital heart disease. He would be disappointed. The book is not a review, but is a paper describing one technique (and not a very good technique). What is described depends on the fact that if radioactive particles are injected intravenously, they normally lodge in the lung; those that end up elsewhere represent a right to left shunt the size of which can be calculated from the relative number of particles. The method described assembles a series of pictures using a gamma-camera from which the data are calculated. I found it difficult to understand how allowance is made for overlaps of pictures and no mention is made of scatter from one view to another or varying softtissue attenuation. However, correlations with results obtained at catheterization are good, and undoubtedly shunts can be assessed by the methods described. The implication of the text suggests that catheterization and angiography can be avoided, but manifestly this is not so. I think if this fairly simple technique were presented as a baseline observation and for follow-up after surgery, then the work would be more realistic. It still does not seem to me to deserve being turned into a monograph. J. P. LAVENDER
96
The Accessory Digestive Organs. Edited by R. E. Wise and A. P. O'Keeffe, pp. xi + 331 (illus), 1975 (Year Book Medical Publishers), £15-40. This book, part of the series sponsored by the American College of Radiologists, is divided intofiveparts: (1) the pancreas; (2) the liver; (3) the extra-hepatic bile-ducts; (4) the gall-bladder; (5) the salivary glands. Each section consists of a number of case reports, with accompanying radiographs, preceded by a short description of the pathology found at that site. The radiographs are of superb quality, and illustrate clearly the varying points which one must consider in reaching a correct diagnosis. The author's use of the term "proper hepatic artery" is, however, both old-fashioned and likely to lead to confusion. The references are practically all from the American literature, and one might finally question the need, other than for illustrative purposes, of invasive investigation of such overt pathology. The book is expensive and will be of use only to a very limited audience. J. A. GLEESON.
(3) A plexiform type where the sinus is composed of a multitude of small veins with rich anastomoses. The lumen of the sinus is traversed by trabeculae giving it the appearance of spongy tissue. Although the right cavernous sinus in this patient appears to consist of multiple channels, the calcification demonstrated within it must be considered as being pathological. Haemangiomata and varices in the orbit are well described (Viallemonte et al., 1959). In some patients a venous malformation may extend outside the orbit into the middle fossa (Hanafee, Sing and Dayton, 1968), through an enlarged sphenoidal fissure (Brauston and Norton, 1963), onto the scalp, frontal bone or hard palate (Lloyd, Wright and Norman, 1971). Lloyd (1974) also describes two patients in whom a haemangioma within the muscle cone was associated with abnormal veins remote from the site of the haemangioma. In both patients these veins lay on the lateral orbital wall. The patient being described showed no evidence of a venous abnormality within the orbit and there was no evidence of an anomaly elsewhere in the skull or face. The condition at the present time is stable and surgery is not contemplated in the immediate future.
CONCLUSIONS
A hitherto undescribed entity of a venous malformation of the right cavernous sinus is described in a 14-year-old boy with proptosis and evidence of involvement of the right sympathetic and third and sixth cranial nerves. The lesion is confined to the cavernous sinus and there is no evidence of a concomitant intra- or extra-orbital venous malformation or of an intra-cranial arterio-venous anomaly. ACKNOWLEDGMENT
We are indebted to Dr. G. Lloyd for his valuable opinion on the radiological changes in this case.
REFERENCES BRAUSTON, B. B., and NORTON, E. N. D., 1963. Intermittent
exophthalmos. American Journal of Ophthalmology, 55, 701-708. HANAFEE, W. N., SING, P. C , and DAYTON, G. O., 1968.
Orbital venography. American Journal of Roentgenology, 104, 29-35. LLOYD, G. A. S., WRIGHT, J. E., and MORGAN, G., 1971.
Venous malformation in the orbit. British Journal of Ophthalmology, 55, 505-516. LLOYD, G. A. S., 1974. Pathological veins in the orbit. British Journal ofRadiology, 47, 570-578. VIALLEFONT, H., PALEIRAC, R., and BOUDET, C , 1959.
The radiological examination of the cavernous sinuses. Bulletin de la Societe Franc Opthalmology 72, 200-219.
Renal cortical necrosis and papillary necrosis in an infant By M. R. Funston, M.B.B.Ch., D.M.R.D., B. J. Cremin, F.R.A.C.R., F.R.C.R., and I. J. K. Tidbury M.B.Ch.B. Department of Radiology, Groote Schuur Hospital, Observatory 7925, Cape Town, So uth Africa {Submitted March, 1975 and in revised form May, 1975)
Bilateral renal cortical necrosis (BRCN) and renal papillary necrosis have been reported in infancy as a complication of gastro-enteritis, perinatal asphyxia, utero-placental haemorrhage, septicaemia, exsanguination, kernicterus, eclampsia, renal vein thrombosis, fetomaternal transfusion and anoxic shock (Mauer and Nogrady, 1969; Leonidas, Berdon and Gribetz, 1971). We wish to report a case of combined BRCN and papillary necrosis, with some unusual radiological findings. Case history A female infant aged three months presented with a fourday history of severe gastro-enteritis followed by convulsions. She was oliguric with metabolic acidosis. The urine contained protein and red cells. The blood urea was 184 and
the serum creatinine 2-5 mg/100 ml. The patient had a spontaneous diuresis without requiring dialysis. In the diuretic phase fluid, electrolyte and acid-base balance was corrected with difficulty. Four weeks after the onset of the illness, an excretory urogram was performed. At the time of the examination the infant was well hydrated, polyuric, and 20 ml. of sodium iothalamate 54 per cent W/V (Conray 325) was injected intravenously. No calcification was present on the control film of the abdomen. Both kidneys were enlarged, measuring 7-5 cm in length. Prompt opacification of the calyces was seen, and all the calyces were noted to be dilated. The ureters were of normal calibre and there was no ureteric or bladder outlet obstruction. A striking feature was dense opacification of the renal cortex and the columns of Bertin, which occurred five minutes after injection of the contrast. The medullary zones were distinctly less opacified. This difference in opacification of the cortex and medulla persisted for 30 minutes. After one hour there was a steady increase in overall kidney density, with equal opacification of the cortex and medulla, which persisted for four days (Fig. 1).
94
FIG. 1. Excretory urogram (four-day film) showing enlargement of the kidneys and a prolonged dense nephrogram.
At the time of this excretory urogram the patient was in the diuretic phase of acute renal failure with a blood urea of 205 mg/100 ml., and there was no reduction in urine output during or after urography. A retrograde uretero-pyelogram showed that the ureters were of normal calibre but bilateral caliectasis was again demonstrated. The absence of ureteric or bladder outlet obstruction was confirmed. Nine weeks after the onset of the illness, bilateral "tramline" cortical calcification, with calcification of the columns of Bertin, was observed on a plain radiograph of the abdomen (Fig. 2). This calcification was only demonstrated with the patient prone and using a low kilovoltage technique. The calcification had completely disappeared 12 weeks later, and a repeat excretory urogram at this stage revealed the typical calyceal changes of papillary necrosis. There was a marked reduction in renal size when compared with the initial urogram. At this time the patient's renal function had returned to normal.
FIG. 2. LogEtronic print of a radiograph of the upper abdomen showing cortical calcification of the right kidney.
The dilatation of the calyces without evidence of obstruction observed in the initial urogram in our patient was considered to be due to swelling of the papillae as a result of papillary necrosis. Changes observed in the subsequent urogram were typical of papillary necrosis, with evidence of sloughed papillae and a reduction in size. Chrispin et al. (1970) and Chrispin (1972) reported heavy opacification by contrast medium of the medullary zones in medullary necrosis. A uniformly dense nephrogram soon after the injection of contrast medium has been reported in patients with acute tubular necrosis by Fry and Cattell (1972). This phenomenon is thought to be the result of leakage of contrast medium into the interstitial space of the kidney. In our patient, however, the heavy opacification occurred in the cortex and the septal columns of Bertin, while the medullary zones appeared relatively radio-translucent. This appearance has not been previously reported. One can only postulate that the degree of tubular damage in the cortex was greater than in the medulla, allowing a greater amount of leakage to occur into the interstitial space of the cortex. The prolonged dense nephrogram, despite prompt calyceal filling, observed in our patient is thought to be the result of the same process, viz. leakage into the interstitial space. Berdon et al. (1969) have reported similar findings. These authors speculated
DISCUSSION
There are numerous reports in the literature of bilateral renal cortical necrosis in infancy (Leonidas et al, 1971; Eskeland and Skogrand, 1959; Mauer and Nogrady, 1969), but the radiographic finding of cortical calcification following this disease in infancy has rarely been observed. Leonidas et al. (1971) and Whelan, Ling and Davis (1967) reported cases of bilateral renal cortical necrosis in infancy followed by cortical calcification. The cortical calcification observed in our patient was not visible on a radiograph exposed with the usual radiographic factors employed for a plain film of the abdomen in an infant. It was only demonstrated using a low kilovoltage technique, with the patient in the prone position, and was not observed in the radiograph taken three months later. We speculate, therefore, that there may be two reasons why this calcification is not more frequently reported in infancy: optimum radiographic factors are not employed, and the calcification may often be temporary. 95
VOL.
49, No. 577 Case reports
that Tamm-Horsfall protein has been precipitated by contrast medium, resulting in tubular blockage and producing a uniformly prolonged tubular nephrogram. However, all their patients were oliguric at the time of excretory urography, whereas our patient was well-hydrated and polyuric. It is therefore unlikely that protein-precipitation was the cause for the prolonged nephrogram in our patient.
CHRISPIN, A. R., 1972. Medullary necrosis in infancy. British Medical Bulletin, 28, 233-236.
ACKNOWLEDGMENTS
LEONIDAS, J. C , BERDON, W. E., and GRIBETZ, D., 1971.
CHRISPIN, A. R., HULL, D., LILLIE, J. G., and RIDSON,
R. A., 1970. Renal tubular necrosis and papillary necrosis after gastro-enteritis in infants. British Medical journal, 1, 410-412. ESKELAND, G., and SKOGRAND, A., 1959. Bilateral cortical
necrosis of the kidneys in infancy. Acta Paediatrica, 48, 278-286. FRY, I. K., and CATTELL, W. R., 1972. The nephrographic
pattern during excretion urography. British Medical Bulletin, 28, 227-232. Bilateral renal cortical necrosis in the newborn infant: Roentgenographic diagnosis. The Journal of Paediatrics, 79, 623-627.
This case was examined during a visit of Dr. W. Berdon of Columbia University and Dr. G. Friedland of the Bowman Grey School of Medicine. They were both most helpful in corroborating our diagnosis, and we are grateful to Dr. Friedland for his suggestion of taking the prone low kilovoltage film, and his help in acquiring the LogEtronic reproduction of Fig. 2.
MCALLISTER, W. H., and NEDELMAN, S. H., 1961.
The
Roentgen manifestations of bilateral renal cortical necrosis. American Journal of Roentgenology, 86,129. MAUER, S. M., and NOGRADY, M. B., 1969. Renal papillary
and cortical necrosis in a newborn infant: Report of a survivor with roentgenolic documentation. The Journal of Paediatrics, 14, 750-754.
REFERENCES BERDON, W. E., SCHWARTZ, R. H., BECKER, J., and BAKER,
D. H., 1969. Tamm-Horsfall proteinuria. Its relationship to prolonged nephrogram in infants and children and to renal failure following intravenous urography in adults with multiple myeloma. Radiology, 92, 714-722.
WHELAN, J. G., LING, J. T., and
DAVIS, L. A.,
1967.
Antemortem Roentgen manifestations of bilateral renal cortical necrosis. Radiology, 89, 682-689.
Book reviews Radionuclide Scanning in Cyanotic Heart Disease. By Gary F. Gates, M.D., pp. xi +100,1974. (Charles C. Thomas, Illinois, USA), $15.00. The earnest reader who sees the title of this book might be misled into thinking that he was about to possess a monograph covering a subject: the use of radionuclides as a noninvasive way of investigating cyanotic congenital heart disease. He would be disappointed. The book is not a review, but is a paper describing one technique (and not a very good technique). What is described depends on the fact that if radioactive particles are injected intravenously, they normally lodge in the lung; those that end up elsewhere represent a right to left shunt the size of which can be calculated from the relative number of particles. The method described assembles a series of pictures using a gamma-camera from which the data are calculated. I found it difficult to understand how allowance is made for overlaps of pictures and no mention is made of scatter from one view to another or varying softtissue attenuation. However, correlations with results obtained at catheterization are good, and undoubtedly shunts can be assessed by the methods described. The implication of the text suggests that catheterization and angiography can be avoided, but manifestly this is not so. I think if this fairly simple technique were presented as a baseline observation and for follow-up after surgery, then the work would be more realistic. It still does not seem to me to deserve being turned into a monograph. J. P. LAVENDER
96
The Accessory Digestive Organs. Edited by R. E. Wise and A. P. O'Keeffe, pp. xi + 331 (illus), 1975 (Year Book Medical Publishers), £15-40. This book, part of the series sponsored by the American College of Radiologists, is divided intofiveparts: (1) the pancreas; (2) the liver; (3) the extra-hepatic bile-ducts; (4) the gall-bladder; (5) the salivary glands. Each section consists of a number of case reports, with accompanying radiographs, preceded by a short description of the pathology found at that site. The radiographs are of superb quality, and illustrate clearly the varying points which one must consider in reaching a correct diagnosis. The author's use of the term "proper hepatic artery" is, however, both old-fashioned and likely to lead to confusion. The references are practically all from the American literature, and one might finally question the need, other than for illustrative purposes, of invasive investigation of such overt pathology. The book is expensive and will be of use only to a very limited audience. J. A. GLEESON.
