Journal of the Royal Society of Medicine Volume 83 March 1990
185
Case reports
Dangerous synergism between sedatives and a hyponatraemic state
F J Lichtigfeld BSc FFPsych
M A Gillman Msc Dsc
South African Brain Research Institute, 173 Louis Botha Avenue, Orange Grove, 2192 Johannesburg, South Africa Keywords: hyponatraemia; electrolyte disturbance; delirium; coma
Hyponatraemia can have deleterious effects on brain function which can be aggravated by various associated conditions'. Indeed it has been suggested that the delirium of electrolyte disturbance is a contraindication to the use of 'large doses of tranquilisers', since the correction of the electrolyte inbalance produces rapid recovery, where no organic damage has occurred2. We report on a case in which an hyponatraemic state may have been aggravated by a combination of low dose sedative medication given on a single occasion to counteract insomnia in a patient whose hyponatremia was only recognized the following day. Such synergism would add another factor not generally recognized1 that could aggravate underlying hyponatraemia to the point of coma and respiratory arrest. This possibly dangerous synergism may be a great deal more common than generally realized. Case report A Caucasian woman of 64 years was admitted to hospital after a gradual deterioration of mental state following the death of her husband some months previously. Since she had suffered insomnia for some weeks and had not responded to previous sedation given on an outpatient basis elsewhere, she was given a combination of promethazine 100 mg, trimipramine 75 mg and clonazepam 2 mg on the night of her admission. She was at the same time given an intramuscular injection of depot flupenthixol 20 mg.- The
Malignant change in rectal duplication
R G Springall ChM FRCS Institute of Child Health, 30 Guildford Street, London WC1N lEW J D Griffiths MS FRCS Royal Marsden Hospital, Fulham Road, London SW3 Keywords: rectum; abnormalities; carcinoma; fistula; colon embryology
Squamous cell carcinoma has been reported in the heterotopic lining of the blind end of a duplication of the
flupenthixol was added as an initial treatment for a suspected severe endogenous depression presenting as a pseudodementia. After this treatment she appeared to settle down well and went to sleep. However at approximately 0300 h next morning her breathing suddenly changed and became laboured. On investigation no apparent mechanical obstruction was found on suction, but she could not be aroused over the next 4 h. To eliminate the possibility of a silent cardiac infarct an ECG was performed, which did not reveal acute pathology. Electrolytes were then measured showing the presence of sodium at 120 meq/l. A consultant physician prescribed oral sodium and that night no further sedation was given; the patient sleeping without such medication. Twenty four hours later the serum sodium appeared normal (140 meq/l) and the patient was much improved in mental alertness. Discussion
This case highlights the importance of serum sodium levels in the diagnostic assessment of any acutely confused patient and further suggests the possibility that there could be a very deleterious synergism existing between an hyponatremic condition and sedative use. Hyponatraemia can often be mild and transient and can thus pass unnoticed or be ignored3. However the delirium of electrolyte disturbance is often associated with sudden changes of electrolyte concentration2, thereby highlighting the importance of the change in level rather than the actual value nieasured as being critical for clinically important sequelae. References 1 Ayus JC, Krothapalli RK, Arieff Al. Treatment of symptomatic hyponatremia and its relation to brain damage. N Engl J Med 1987;317:1190-5 2 Oakely HF. Psychiatric emergencies in endocrine and metabolic disease. In: Sonksen PH, Lowy C, eds. Clinics in endocrinology and metabolism, vol 9. London: WB Saunders 1980;615-24 3 Gill GV, Flear CTG. Hyponatraemia, In: Price CP, Alberti KGMM, eds Recent advances in clinical biochemistry no 3. London: Churchill Livingstone, 1985:149-76 (Accepted 10 January 1989)
0141-0768/90/ 030185-01/$02.00/0 © 1990 The Royal Society of Medicine
ascending and transverse colon'. Adenocarcinoma in a villous adenoma was recorded in a duplication arising from the ascending colon2. Reports of malignant change in a separate duplication cyst rather than a segment in continuity with the bowel have appeared in the literature3.
Case report A male aged 49 presented with acute retention of urine, following one year of lower back pain and 6 months of increasing stool frequency. A hard mass was palpable per rectum posteriorly, indenting but not invading the bowel. Intravenous urography demonstrated the 15 cm mass within the pelvis. The bladder was displaced upwards and anteriorly and evacuated poorly. Needle biopsy was inconclusive and the lesion needed decompression. Through a horizontal post-anal incision 500 ml of infected gelatinous fluid were evacuated. Histology was again
0141-0768/90/ 030185-03/$02.00/0 © 1990 The Royal Society of Medicine
186
Journal of the Royal Society of Medicine Volume 83 March 1990 The resected specimen demonstrated an inflammatory mass lying adjacent to the bowel, surrounding a fistula track, partially lined by well differentiated mucous adenocarcinoma (Figure 2).
FIBROUS EXTENSION
FISTULA
DUPLICATED RECTAL STUMP
Figure 1. Representation of rectal duplication
3 2
.. ..... ...
.. ......
J.
T.
Mr
ROM
Fiagure 2. Resected specimen non-specific. Bacteriology demonstrated multiple bowel organisms. He developed a faecal fistula through the perineal wound which a sinogram failed to trace. Because of the persistent faecal loss, proximal colostomy was performed. Sigmoidoscopy had shown an area of granulation tissue at 14 cm which at operation coincided with a fistula communicating with a retro-rectal cavity filled with faecal material and surrounded by inflammatory tissue. A diagnosis of rectal reduplication was made and confirmed on biopsy. The perineal sinus continued to drain pus and mucus and a Hartmann procedure was performed one year later with the rectal reduplication left in situ. The patient remained well for the next 12 years until he presented with buttock pain. A perianal abscess was surgically drained. A further cavity was then discovered tracking above the pelvic floor muscles which contained clear mucinous material. Multiple biopsy specimens demonstrated adenocarcinoma within the non-infected cavity. This was a cancer developing within the rectal reduplication penetrating the levator anti lateral to the rectum. Abdomino-perineal excision of the rectal remnant was performed. The duplication communicated distally with the functioning bowel. Above, the lumen became obliterated and continued as a dense fibrous core merging with the retro peritoneal tissue surrounding the great vessels (Figure 1).
Discussion In 1984 Fitz4 coined the term duplication for the developmental anomaly in which a double segment of the bowel is represented by a cyst, diverticulum or full length colonic duplication. In experimental animals, hypoxia or trauma to the fetus can induce duplication or even twinning'6. The original postulate for the embryogenesis of duplications was put forward by Lewis and Thyng7 who thought their origin lay in cell nest islands. Bremers8 theory of incomplete resolution ofthe 'solid phase' of intestinal development was discredited by Johnson9 who showed that the oesophagus and colon never undergo a 'solid plug' stage upon which the vacuolation theory depends. Duplication of the alimentary tract is most common in the oesophagus and ileum but may occur from tongue to rectum10. They may have a single opening to the bowel but sometimes recommunicate distally. Eighty per cent of rectal duplications do not join with the bowel lumen11. Duplication of the colon is divided into three different types. Mesenteric cysts within the mesentery of the colon or rectum, frequently share a common blood supply, are lined by intestinal epithelium and have a muscular wall. At the other extreme is long duplication, frequently involving replication ofthe entire colon or rectum. They may terminate in separate anal canals or the inner loop may end at an ectopic site on the perineum, urethra or vagina. The duplicated anus is frequently imperforate and unsuspected from the exterior. Colonic diverticula frequently share a common wall through part of their length and have a shared blood supply. They are lined with intestinal mucosa which may be inappropriate for their site of origin12. Duplications, being congenital lesions, most commonly present in childhood. Barium enema can demonstrate a communication between the duplication and the normal bowel or may just show a filling defect. Treatment is usually by segmental bowel excision together with the associated cyst. If complete resection is not feasible then partial resection and internal drainage can be accomplished to reduce the risk of impacting the distal part of the anomaly. When tubular duplications are identified in the colon, patients should be investigated for possible urinary tract
malformations13. The age ofthis patient at presentation was one ofthe oldest reported"4. In the past the consensus has been to treat these congenital bowel malformations as harmless and to manage them conservatively to avoid damage to associated structures. This report demonstrates the need for caution in such a policy, particularly when the redundant segment is not open to visualization. It is suggested that where a low anomaly exists the correct management is wide excision, utilizing a pull through procedure or even an abdominoperineal approach. References 1 Larizadeh R, Powell DEB. Neoplastic change in a duplicated colon. Br J Surg 1965;52:666-8 2 Tamaney HJ, Testa RE. Carcinoma arising in a duplicated colon. Cancer 1967;20:478-81 3 Gibson TC, Edwards JM, Strafig S. Carcinoma arising in a rectal duplication cyst. Br J Surg 1986;73:377 4 Fitz RH. Persistent omphalo-mesenteric remnant. Am J Med
1884;88:30-57 5
6 7
8 9
Inlgalls TH,
Philbrook FR. Monstrosities induced by hypoxia. N Engl J Med 1958~,269:558-64 Louw JH. Congenital intestinal atresia and stenosis in the newborn. Ann R Coil Surg Engl 1959;,2C:209-34 LewiFT, Thyng FW. Regular occurrece of intestinal divrerticula inembryos ofthe pig, rabbit and man. Am JAnat 1907;7:505-19 Bremer JL. Diverticula and duplications of the intestinal tract. Arch Pat/wi 1944;38:132-40 Johnson FP. Development of the mucous membrane of the large intestine and vermiform process in the human embryo. Am J Anat 1913;14:187-233
Journal of the Royal Society of Medicine Volume 83 March 1990 10 Wilkinson DJ, Wilkinson KW, Hojdun N. Intestinal duplication. A report of two cases. Br J Radiology 1973;46: 1070-2 11 Gross RE, Holcombe GW, Farber S. Duplications of the alimentary tract. Pediatrics 1952;9:449-67 12 Mellish RPW, Koop CE. Clinical manifestations of duplication of the bowel. Pediatrics 1961;27:397-407
The shrinking seminoma
A H R W Simpson FRCSa D G Calvert FRCSa B W Codling MD FRCPathb Departments of aGeneral Surgery and bHistopathology, Gloucestershire Royal Hospital, Gloucester Keywords: seminoma; presentation
It is almost invariable that 'solid' neoplasms present as swellings or enlargements'-3. If, however, such a lesion were to replace normal tissue, and then undergo necrosis, it could lead to the host organ actually becoming smaller at the time of presentation. Such a case is reported here, and illustrates that, in the testis at least, any change in size must be considered with suspicion and investigated. Case report An otherwise fit 43-year-old man presented with a one year history of his left testicle becoming smaller than that on the right. This had been associated with vague intermittent pains which were never severe enough to suggest an acute torsion. There was no history of trauma or infection (eg mumps or penile discharge). On examination, the left testicle was approximately 30% smaller than on the right. It was harder and rounder, and compression produced a different sensation to that on the other side. The patient was otherwise totally fit. Preoperative
187
13 Ravitch MM, Scott WW. Duplications ofthe entire colon, bladder and urethra. Surgery 1953;34:843-58 14 Weitzel RA, Breed JR. Carcinoma arising in a rectal duplication (Enterocystoma) Am Surg 1963;157:476-80
(Accepted 17 January 1989. Correspondence to R G Springall, 63 Cloudesley Road, Barnsbury, London Nl OEL)
investigations such as serum human chorionic gonadotrophin (HCG) and alpha fetoprotein (AFP) as well as a lymphangiogram and an abdominal CT scan were all normal. At operation, the appearance was that of a tumour and therefore an orchidectomy was performed. The testis measured 4 x 3 x 3 cm and the cut surface revealed a partly haemorrhagic tumour of variable colour measuring 3x3x2 cm. Histological examination (Figure 1) revealed a seminoma with extensive areas of necrosis. The viable areas showed the typical microscopic pattern of a seminoma with a moderate lymphocytic infiltrate. Multiple sections failed to reveal any teratomatous, trophoblastic or yolk sac elements. The tumour had invaded locally. In addition the remaining testicular tissue was markedly atrophic although there was a small amount of fibrosis. Postoperatively, the patient made an uneventful recovery. He received a course of radiotherapy (3000 rad - anterior and posterior dogleg and 3000 rad direct to scrotum). He is being followed closely and his AFP and HCG are checked regularly and remain normal. He is well and symptom free 18 months after presentation. Discussion Atrophy of the testis is reported as a result of ischaemic episodes due to intermittent torsion4. It has also occurred with inflammatory and endocine disease. However sinkage of the testis containing a tumour must be exceedingly uncommon, and we have been unable to find any similar documented report, although this is undoubtedly what happened in this case. The mechanism must be presumed to have been neoplastic growth replacing normal tissue with associated pressure atrophy, followed by episodes of tumour necrosis; this would explain the episodes of pain experienced by the patient. Ultrasonography would have shown the abnormal architecture of the testis but it is unlikely that it would be able to distinguish between a neoplastic or ischaemic cause for the atrophy. Unless the clinician is aware ofthis mode of presentation he may consider that the testis is suffering from intermittent torsion rather than a tumour and he may thus approach the testis transcrotally. Such massive necrosis of a tumour is by no means uncommon in other germ cell neoplasms, but is unusual in a pure seminoma. The pathologist must sample specimens from similar patients extensively to exclude any atypical elements. In the present case there was no histological or serological evidence of non-seminomatous elements. Nevertheless, whatever the exact pathological nature, the clinician should be aware that any change in size of a testicle must be regarded with suspicion, and that shrinkage as well as enlargement necessitates fill investigation for malignancy. Acknowledgments The authors are grateful to Dr R H W Simpson for his advice. References 1 Blandy JP, Hope-Stone HF, Dayan AD. The clinical features of neoplasms of the testicle. In: Tumour8 of the testicle. London: Heinemann, 1970:56-66 2 Stephen RA. The clinical presentation of testicular tumours. Br J Urol 1962;34:448-50 3 Thompson IM, Wear J, Almond C, Schewe DJ, Sala J. An analytical survey of 178 testicular tumors. J Urol 1961;85:773-9 4 Creagh TA, McDermott TE, McLean PA, Walsh A. Intermittent torsion of the testis. Br Med J 1988,27:525-6
Figure 1. A section ofthe specimen stained with elastic van Giesen. Magnificationx3
(Accepted 23 January 1989. Correspondence to Mr A H R WSimpson, The Nuffield Orthopaedic Centre, Oxford)
0141-0768/90/ 030187-01/02.00/0 © 1990 The Royal Society of Medicine
185
Case reports
Dangerous synergism between sedatives and a hyponatraemic state
F J Lichtigfeld BSc FFPsych
M A Gillman Msc Dsc
South African Brain Research Institute, 173 Louis Botha Avenue, Orange Grove, 2192 Johannesburg, South Africa Keywords: hyponatraemia; electrolyte disturbance; delirium; coma
Hyponatraemia can have deleterious effects on brain function which can be aggravated by various associated conditions'. Indeed it has been suggested that the delirium of electrolyte disturbance is a contraindication to the use of 'large doses of tranquilisers', since the correction of the electrolyte inbalance produces rapid recovery, where no organic damage has occurred2. We report on a case in which an hyponatraemic state may have been aggravated by a combination of low dose sedative medication given on a single occasion to counteract insomnia in a patient whose hyponatremia was only recognized the following day. Such synergism would add another factor not generally recognized1 that could aggravate underlying hyponatraemia to the point of coma and respiratory arrest. This possibly dangerous synergism may be a great deal more common than generally realized. Case report A Caucasian woman of 64 years was admitted to hospital after a gradual deterioration of mental state following the death of her husband some months previously. Since she had suffered insomnia for some weeks and had not responded to previous sedation given on an outpatient basis elsewhere, she was given a combination of promethazine 100 mg, trimipramine 75 mg and clonazepam 2 mg on the night of her admission. She was at the same time given an intramuscular injection of depot flupenthixol 20 mg.- The
Malignant change in rectal duplication
R G Springall ChM FRCS Institute of Child Health, 30 Guildford Street, London WC1N lEW J D Griffiths MS FRCS Royal Marsden Hospital, Fulham Road, London SW3 Keywords: rectum; abnormalities; carcinoma; fistula; colon embryology
Squamous cell carcinoma has been reported in the heterotopic lining of the blind end of a duplication of the
flupenthixol was added as an initial treatment for a suspected severe endogenous depression presenting as a pseudodementia. After this treatment she appeared to settle down well and went to sleep. However at approximately 0300 h next morning her breathing suddenly changed and became laboured. On investigation no apparent mechanical obstruction was found on suction, but she could not be aroused over the next 4 h. To eliminate the possibility of a silent cardiac infarct an ECG was performed, which did not reveal acute pathology. Electrolytes were then measured showing the presence of sodium at 120 meq/l. A consultant physician prescribed oral sodium and that night no further sedation was given; the patient sleeping without such medication. Twenty four hours later the serum sodium appeared normal (140 meq/l) and the patient was much improved in mental alertness. Discussion
This case highlights the importance of serum sodium levels in the diagnostic assessment of any acutely confused patient and further suggests the possibility that there could be a very deleterious synergism existing between an hyponatremic condition and sedative use. Hyponatraemia can often be mild and transient and can thus pass unnoticed or be ignored3. However the delirium of electrolyte disturbance is often associated with sudden changes of electrolyte concentration2, thereby highlighting the importance of the change in level rather than the actual value nieasured as being critical for clinically important sequelae. References 1 Ayus JC, Krothapalli RK, Arieff Al. Treatment of symptomatic hyponatremia and its relation to brain damage. N Engl J Med 1987;317:1190-5 2 Oakely HF. Psychiatric emergencies in endocrine and metabolic disease. In: Sonksen PH, Lowy C, eds. Clinics in endocrinology and metabolism, vol 9. London: WB Saunders 1980;615-24 3 Gill GV, Flear CTG. Hyponatraemia, In: Price CP, Alberti KGMM, eds Recent advances in clinical biochemistry no 3. London: Churchill Livingstone, 1985:149-76 (Accepted 10 January 1989)
0141-0768/90/ 030185-01/$02.00/0 © 1990 The Royal Society of Medicine
ascending and transverse colon'. Adenocarcinoma in a villous adenoma was recorded in a duplication arising from the ascending colon2. Reports of malignant change in a separate duplication cyst rather than a segment in continuity with the bowel have appeared in the literature3.
Case report A male aged 49 presented with acute retention of urine, following one year of lower back pain and 6 months of increasing stool frequency. A hard mass was palpable per rectum posteriorly, indenting but not invading the bowel. Intravenous urography demonstrated the 15 cm mass within the pelvis. The bladder was displaced upwards and anteriorly and evacuated poorly. Needle biopsy was inconclusive and the lesion needed decompression. Through a horizontal post-anal incision 500 ml of infected gelatinous fluid were evacuated. Histology was again
0141-0768/90/ 030185-03/$02.00/0 © 1990 The Royal Society of Medicine
186
Journal of the Royal Society of Medicine Volume 83 March 1990 The resected specimen demonstrated an inflammatory mass lying adjacent to the bowel, surrounding a fistula track, partially lined by well differentiated mucous adenocarcinoma (Figure 2).
FIBROUS EXTENSION
FISTULA
DUPLICATED RECTAL STUMP
Figure 1. Representation of rectal duplication
3 2
.. ..... ...
.. ......
J.
T.
Mr
ROM
Fiagure 2. Resected specimen non-specific. Bacteriology demonstrated multiple bowel organisms. He developed a faecal fistula through the perineal wound which a sinogram failed to trace. Because of the persistent faecal loss, proximal colostomy was performed. Sigmoidoscopy had shown an area of granulation tissue at 14 cm which at operation coincided with a fistula communicating with a retro-rectal cavity filled with faecal material and surrounded by inflammatory tissue. A diagnosis of rectal reduplication was made and confirmed on biopsy. The perineal sinus continued to drain pus and mucus and a Hartmann procedure was performed one year later with the rectal reduplication left in situ. The patient remained well for the next 12 years until he presented with buttock pain. A perianal abscess was surgically drained. A further cavity was then discovered tracking above the pelvic floor muscles which contained clear mucinous material. Multiple biopsy specimens demonstrated adenocarcinoma within the non-infected cavity. This was a cancer developing within the rectal reduplication penetrating the levator anti lateral to the rectum. Abdomino-perineal excision of the rectal remnant was performed. The duplication communicated distally with the functioning bowel. Above, the lumen became obliterated and continued as a dense fibrous core merging with the retro peritoneal tissue surrounding the great vessels (Figure 1).
Discussion In 1984 Fitz4 coined the term duplication for the developmental anomaly in which a double segment of the bowel is represented by a cyst, diverticulum or full length colonic duplication. In experimental animals, hypoxia or trauma to the fetus can induce duplication or even twinning'6. The original postulate for the embryogenesis of duplications was put forward by Lewis and Thyng7 who thought their origin lay in cell nest islands. Bremers8 theory of incomplete resolution ofthe 'solid phase' of intestinal development was discredited by Johnson9 who showed that the oesophagus and colon never undergo a 'solid plug' stage upon which the vacuolation theory depends. Duplication of the alimentary tract is most common in the oesophagus and ileum but may occur from tongue to rectum10. They may have a single opening to the bowel but sometimes recommunicate distally. Eighty per cent of rectal duplications do not join with the bowel lumen11. Duplication of the colon is divided into three different types. Mesenteric cysts within the mesentery of the colon or rectum, frequently share a common blood supply, are lined by intestinal epithelium and have a muscular wall. At the other extreme is long duplication, frequently involving replication ofthe entire colon or rectum. They may terminate in separate anal canals or the inner loop may end at an ectopic site on the perineum, urethra or vagina. The duplicated anus is frequently imperforate and unsuspected from the exterior. Colonic diverticula frequently share a common wall through part of their length and have a shared blood supply. They are lined with intestinal mucosa which may be inappropriate for their site of origin12. Duplications, being congenital lesions, most commonly present in childhood. Barium enema can demonstrate a communication between the duplication and the normal bowel or may just show a filling defect. Treatment is usually by segmental bowel excision together with the associated cyst. If complete resection is not feasible then partial resection and internal drainage can be accomplished to reduce the risk of impacting the distal part of the anomaly. When tubular duplications are identified in the colon, patients should be investigated for possible urinary tract
malformations13. The age ofthis patient at presentation was one ofthe oldest reported"4. In the past the consensus has been to treat these congenital bowel malformations as harmless and to manage them conservatively to avoid damage to associated structures. This report demonstrates the need for caution in such a policy, particularly when the redundant segment is not open to visualization. It is suggested that where a low anomaly exists the correct management is wide excision, utilizing a pull through procedure or even an abdominoperineal approach. References 1 Larizadeh R, Powell DEB. Neoplastic change in a duplicated colon. Br J Surg 1965;52:666-8 2 Tamaney HJ, Testa RE. Carcinoma arising in a duplicated colon. Cancer 1967;20:478-81 3 Gibson TC, Edwards JM, Strafig S. Carcinoma arising in a rectal duplication cyst. Br J Surg 1986;73:377 4 Fitz RH. Persistent omphalo-mesenteric remnant. Am J Med
1884;88:30-57 5
6 7
8 9
Inlgalls TH,
Philbrook FR. Monstrosities induced by hypoxia. N Engl J Med 1958~,269:558-64 Louw JH. Congenital intestinal atresia and stenosis in the newborn. Ann R Coil Surg Engl 1959;,2C:209-34 LewiFT, Thyng FW. Regular occurrece of intestinal divrerticula inembryos ofthe pig, rabbit and man. Am JAnat 1907;7:505-19 Bremer JL. Diverticula and duplications of the intestinal tract. Arch Pat/wi 1944;38:132-40 Johnson FP. Development of the mucous membrane of the large intestine and vermiform process in the human embryo. Am J Anat 1913;14:187-233
Journal of the Royal Society of Medicine Volume 83 March 1990 10 Wilkinson DJ, Wilkinson KW, Hojdun N. Intestinal duplication. A report of two cases. Br J Radiology 1973;46: 1070-2 11 Gross RE, Holcombe GW, Farber S. Duplications of the alimentary tract. Pediatrics 1952;9:449-67 12 Mellish RPW, Koop CE. Clinical manifestations of duplication of the bowel. Pediatrics 1961;27:397-407
The shrinking seminoma
A H R W Simpson FRCSa D G Calvert FRCSa B W Codling MD FRCPathb Departments of aGeneral Surgery and bHistopathology, Gloucestershire Royal Hospital, Gloucester Keywords: seminoma; presentation
It is almost invariable that 'solid' neoplasms present as swellings or enlargements'-3. If, however, such a lesion were to replace normal tissue, and then undergo necrosis, it could lead to the host organ actually becoming smaller at the time of presentation. Such a case is reported here, and illustrates that, in the testis at least, any change in size must be considered with suspicion and investigated. Case report An otherwise fit 43-year-old man presented with a one year history of his left testicle becoming smaller than that on the right. This had been associated with vague intermittent pains which were never severe enough to suggest an acute torsion. There was no history of trauma or infection (eg mumps or penile discharge). On examination, the left testicle was approximately 30% smaller than on the right. It was harder and rounder, and compression produced a different sensation to that on the other side. The patient was otherwise totally fit. Preoperative
187
13 Ravitch MM, Scott WW. Duplications ofthe entire colon, bladder and urethra. Surgery 1953;34:843-58 14 Weitzel RA, Breed JR. Carcinoma arising in a rectal duplication (Enterocystoma) Am Surg 1963;157:476-80
(Accepted 17 January 1989. Correspondence to R G Springall, 63 Cloudesley Road, Barnsbury, London Nl OEL)
investigations such as serum human chorionic gonadotrophin (HCG) and alpha fetoprotein (AFP) as well as a lymphangiogram and an abdominal CT scan were all normal. At operation, the appearance was that of a tumour and therefore an orchidectomy was performed. The testis measured 4 x 3 x 3 cm and the cut surface revealed a partly haemorrhagic tumour of variable colour measuring 3x3x2 cm. Histological examination (Figure 1) revealed a seminoma with extensive areas of necrosis. The viable areas showed the typical microscopic pattern of a seminoma with a moderate lymphocytic infiltrate. Multiple sections failed to reveal any teratomatous, trophoblastic or yolk sac elements. The tumour had invaded locally. In addition the remaining testicular tissue was markedly atrophic although there was a small amount of fibrosis. Postoperatively, the patient made an uneventful recovery. He received a course of radiotherapy (3000 rad - anterior and posterior dogleg and 3000 rad direct to scrotum). He is being followed closely and his AFP and HCG are checked regularly and remain normal. He is well and symptom free 18 months after presentation. Discussion Atrophy of the testis is reported as a result of ischaemic episodes due to intermittent torsion4. It has also occurred with inflammatory and endocine disease. However sinkage of the testis containing a tumour must be exceedingly uncommon, and we have been unable to find any similar documented report, although this is undoubtedly what happened in this case. The mechanism must be presumed to have been neoplastic growth replacing normal tissue with associated pressure atrophy, followed by episodes of tumour necrosis; this would explain the episodes of pain experienced by the patient. Ultrasonography would have shown the abnormal architecture of the testis but it is unlikely that it would be able to distinguish between a neoplastic or ischaemic cause for the atrophy. Unless the clinician is aware ofthis mode of presentation he may consider that the testis is suffering from intermittent torsion rather than a tumour and he may thus approach the testis transcrotally. Such massive necrosis of a tumour is by no means uncommon in other germ cell neoplasms, but is unusual in a pure seminoma. The pathologist must sample specimens from similar patients extensively to exclude any atypical elements. In the present case there was no histological or serological evidence of non-seminomatous elements. Nevertheless, whatever the exact pathological nature, the clinician should be aware that any change in size of a testicle must be regarded with suspicion, and that shrinkage as well as enlargement necessitates fill investigation for malignancy. Acknowledgments The authors are grateful to Dr R H W Simpson for his advice. References 1 Blandy JP, Hope-Stone HF, Dayan AD. The clinical features of neoplasms of the testicle. In: Tumour8 of the testicle. London: Heinemann, 1970:56-66 2 Stephen RA. The clinical presentation of testicular tumours. Br J Urol 1962;34:448-50 3 Thompson IM, Wear J, Almond C, Schewe DJ, Sala J. An analytical survey of 178 testicular tumors. J Urol 1961;85:773-9 4 Creagh TA, McDermott TE, McLean PA, Walsh A. Intermittent torsion of the testis. Br Med J 1988,27:525-6
Figure 1. A section ofthe specimen stained with elastic van Giesen. Magnificationx3
(Accepted 23 January 1989. Correspondence to Mr A H R WSimpson, The Nuffield Orthopaedic Centre, Oxford)
0141-0768/90/ 030187-01/02.00/0 © 1990 The Royal Society of Medicine
