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Penile Gangrene Associated with Chronic Renal Failure: Report of 7 Cases and Review of the Literature
0022-5347/94/1526-2014$03.00/0 Vol. 152, 2014-2016, December 1994 Printed in U.S.A.
THE JOURNAL OF UROLOGY Copyright© 1994 by AMERICAN UROLOGICAL AssOCIATION, INC.
PENILE GANGRENE ASSOCIATED WITH CHRONIC RENAL FAILURE: REPORT OF 7 CASES AND REVIEW OF THE LITERATURE MARK STEIN, CHRISTIAN ANDERSON, RICCARDO RICCIARDI, JOE W. CHAMBERLIN, SETH E. LERNER AND DANIEL GLICKLICH From the Departments of Urology and Medicine, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
ABSTRACT
Distal penile gangrene associated with renal failure is a rare entity with only 3 cases reported in the literature. Certain physiological abnormalities are commonly found in association with this condition, including secondary hyperparathyroidism, diabetes and peripheral vascular disease. We report our experience with 7 patients who presented with this condition. All patients had end stage renal disease with 5 on hemodialysis, 1 on peritoneal dialysis and 1 with a functioning cadaveric renal transplant. Six patients had diabetes mellitus and all had derangements of the calcium and phosphate metabolism, with the calcium-phosphorus product being greater than 70. Five patients were treated expectantly with resolution of gangrene in 2 and stable disease in 3. Three of the 5 patients managed expectantly and both patients treated with penectomy died of unrelated causes within 3 months. We conclude that there is no advantage to aggressive surgical treatment of penile gangrene associated with renal failure, since the outcome is the same. The overall mortality for this group is high due to associated co-morbid disease regardless of the type of treatment. Furthermore, subtotal parathyroidectomy is not indicated, since spontaneous improvement and mortality rates reported in our series were similar to those of previously reported cases. Expectant management of affected patients affords the best treatment. KEY WORDS:
penis; gangrene; renal failure, chronic
Distal penile gangrene in patients with chronic renal failure is a rare entity, with only 3 cases in the literature. 1 - 3 Unlike Fournier's gangrene, this disease is limited to the glans penis or distal penile shaft and usually follows a relatively indolent course (see figure). Affected patients frequently suffer from disorders associated with renal failure, such as diabetes mellitus, hypertension and occlusive arterial disease. Distal penile gangrene is believed to be a variant of diffuse, rapidly progressive arterial calcification,4· 5 which is attributed to secondary hyperparathyroidism that occurs in patients with chronic renal failure, and leads to arterial occlusion and distal extremity loss. Historically, treatment has included surgical removal of the parathyroid and extirpation of gangrenous areas. A high associated mortality rate is reported despite this aggressive management. To define further this rare entity and improve upon the management strategy, we reviewed our experience with 7 patients who presented with distal penile gangrene and chronic renal failure during a 4-year period.
hyperparathyroidism consisting of an elevated calcium-phosphorus product. Patients 1 al).d 2 complained of worsening peripheral vascular disease. Patient 1 had rest pain in all 10 fingers as well as dry gangrene of the distal digits and patient 2 had severe left lower extremity ischemia requiring attempted balloon angioplasty of the left popliteal artery followed by left above the knee amputation. Patient 1 had focal gangrenous patches on the glans penis. Due to phimosis, which precluded local wound care, circumcision was performed. Gangrene progressed following circumcision and the patient underwent partial penectomy. The necrotic areas progressed despite local wound care and penectomy with perinea! urethrostomy
PATIENT INFORMATION
Between July 1989 and April 1993, 621 patients 50 to 87 years old (mean age 66.6 years) with chronic renal failure requiring dialysis were treated at our university medical center and 2 affiliated dialysis units. Of these patients urological consultation was requested for 7 to evaluate penile gangrene or penile pain. Detailed patient information is presented in table 1. Of the 7 patients 6 had diabetes mellitus. Six patients were dialysis dependent, while 1 had a functioning cadaveric renal allograft. All patients had clinical evidence of preexisting occlusive vascular disease, some with more than 1 type (5 with peripheral vascular disease, 4 with coronary artery disease and 1 with cerebrovascular disease). All patients had biochemical evidence of secondary Accepted for publication March 25, 1994.
Patient 7. Distal penile gangrene associated with renal failure 2014
2015
PENILE GANGRENE ASSOCIATED WITH CHRONIC RENAL FAILURE TABLE 1.
Pt.-Age No.
Age, dialysis status, medical history and calcium-phosphorus product in patients with penile gangrene Systemic Diseases
Dialysis (mos.)
1-71
Diabetes mellitus, end stage renal disease
Hemodialysis (12)
2-73
Diabetes mellitus, end stage renal disease, colon Ca Diabetes mellitus, myocardial infarction, congestive heart failure, end stage renal disease Intravenous drug abuse, chronic obstructive pulmonary disease, tuberculosis, end stage renal disease Diabetes mellitus, hypertension, congestive heart failure, end stage renal disease, cirrhosis Diabetes mellitus, cerebrovascular accident, myocardial infarction, end stage renal disease Coronary artery disease, diabetes mellitus, chronic obstructive pulmonary disease, end stage renal disease
Hemodialysis (5)
3-54 4-65 5-50 6-87 7-66
Transplant
Prior Vascular Disease
CalciumPhosphorus Product
Above knee amputation, pain and gangrene of digits Above knee amputation
71.5
Below knee amputation, rt. digit amputation, colostomy for ischemic bowel, coronary artery disease
75.9
Peritoneal dialysis (more than 24)
78.3
72.0
Hemodialysis (18)
Coronary artery disease
76.0
Hemodialysis (more than 24)
Lower extremity rest pain
77.3
Hemodialysis (more than 24)
Coronary artery disease
71.0
was later required. The patient died of overwhelming sepsis unrelated to the operation 3 months later. Patient 2 was managed expectantly and the gangrenous area appeared to stabilize. He died of a massive cerebrovascular accident soon after presentation. Two patients had distal penile gangrene following acute hypotensive episodes and myocardial infarction. Patient 3 had dry gangrene of the entire glans penis and was managed expectantly with autoamputation of the gangrenous segment within 4 weeks. Subsequently, cellulitis of the remaining area developed, which was treated with intravenous antibiotics and local wound care. The patient was discharged from the hospital and has had no further morbidity. Patient 4 had focal dry gangrenous patches on the glans penis. He was treated conservatively with topical antibiotic ointment with resolution of the gangrene. This patient died of overwhelming sepsis due to an unrelated cause 1 month later. Three patients presented with recurrent balanitis and phimosis, and were scheduled for elective circumcision. Patient 5 had gangrenous areas along the distal prepuce and glans before the elective circumcision. He was initially treated with circumcision and debridement of the necrotic areas. Subsequently, the devitalized areas progressed and partial penectomy was performed. He was discharged home but rehospitalized 2 months later with a polymicrobial necrotizing fasciitis of the leg and he died. Patients 6 and 7 underwent elective circumcision for phimosis. Subsequently, within 1 week gangrenous patches and total glandular gangrene developed, respectively. Both patients were managed conservatively with local wound care. Patient 6 improved and was discharged home without any further morbidity. The area of gangrene stabilized in patient 7 but he died of multi-organ system failure several months later. DISCUSSION
Diffuse vascular calcification is a manifestation of abnormal calcium and phosphorus metabolism that occurs in patients with chronic renal failure and in renal transplant recipients. 4 - 6 Arterial and metastatic soft tissue calcification occurs as a result of secondary hyperparathyroidism and its associated elevation of the serum calcium-phosphorus product. The risk for metastatic calcification is believed to be considerable when the calcium-phosphorus product exceeds 70. 7 - 9 The etiology of secondary hyperparathyroidism inuremic patients has been well described. 10 Histological examination of affected arteries reveals calcific infiltration of the media with reactive connective tissue hyperplasia of the in-
tima that results in luminal obliterations and ultimately progression to distal tissue gangrene. 3 • 11 There have been several reports in the literature of distal extremity gangrene associated with abnormal calcium metabolism. Gipstein et al reported on 11 patients with end stage renal disease, high or high normal serum calcium levels and severe hyperphosphatemia. 5 Of these patients 10 were treated with parathyroidectomy, of whom 7 had improvement in the ischemic lesions. They concluded that parathyroidectomy is an effective treatment and that early institution of phosphate binding agents may prevent this syndrome. Perloff et al reported on 4 patients with post-transplantation calcinosis, of whom 1 had distal penile gangrene. 4 All patients were treated with subtotal parathyroidectomy but only 1 survived to be discharged from the hospital. They recommended subtotal parathyroidectomy with autotransplantation despite the poor outcome and lack of correlation between the operation and healing of distal gangrenous ulcers. The penile gangrene in 1 patient in this series resulted from a spreading staphylococcal infection from the thigh, similar to Fournier's gangrene, and so this does not represent the type of penile gangrene discussed in our series. Fournier's gangrene is a form of bacterial necrotizing fasciitis that causes wet gangrene. Diffuse vascular calcification results in obliteration of arterial inflow, causing ischemic dry gangrene. Hallgren et al presented 2 cases of progressive arterial calcifications resulting in peripheral gangrene, treated by subtotal parathyroidectomy. 6 Of the 2 patients 1 improved postoperatively. In this case there was no evidence of parathyroid hyperplasia in the pathological specimen. One patient with mild glandular hyperplasia on pathological section died shortly following surgery. Isolated penile gangrene is believed to be a focal manifestation of diffuse arterial calcification seen in chronic renal failure patients. Lowe and Brendler described a patient who was initially managed conservatively until distal penectomy was required for control of ischemic pain. 2 Subtotal parathyroidectomy was performed to stabilize the vascular deterioration. The patient ultimately required amputation of 2 fingers for preexisting gangrene, and a left below the knee amputation for nonhealing foot and ankle ulcers. Ashouri and Perez reported on a case in which the necrotic areas were excised without further patient morbidity. 1 Our report expands the literature of penile gangrene as a result of diffuse arterial calcification associated with chronic renal failure from 3 cases to 10. The mortality rate for this
2016
PENILE GANGRENE ASSOCIATED WITH CHRONIC RENAL FAILURE TABLE
2. Presenting symptoms, clinical findings, treatment, outcome and mortality in patients with penile gangrene
Presenting Signs and Symptoms
Gross Description
Treatment
1
Worsening peripheral vascular disease
Focal gangrenous patches
Circumcision, partial and then total penectomy with perinea! urethrostomy
2
Entire glans
Observation
Entire glans
Observation, autoamputation, antibiotics, debridement Observation
Gangrene of penile tissues
4
Worsening peripheral vascular disease Shock after myocardial infarction Urosepsis, shock
5
Painful phimosis
Gangrene of distal glans, gram-pos. cocci
6
After circumcision
Circumcision, debridement, partial penectomy Observation
Pt. No.
3
Focal gangrenous patches Distal prepuce, glans Focal gangrenous patches Entire glans
Pathology Gangrene of glans and corpus cavernosum, thrombi, calcified small and medium vessels Gangrene of glans
Outcome Died of sepsis*
Died of myocardial infarction* Alive, serum creatinine 1.2 mg./dl.t Died of sepsist Died of sepsis* Alivet
Died of multi-organ Focal gangrene of foreskin system failuret All circumcisions were performed using the sleeve or dorsal slit technique, without the use of a tourniquet. Minimal, superficial cautery was used for hemostasis. * No progression of penile gangrene; died of unrelated causes within 3 months. t No further progression of penile gangrene. t Gangrenous area improved before death. 7
After circumcision
Observation
group remains high due to the severity of the associated systemic illnesses: 5 of the 7 patients died within 3 months after presentation (table 2). This rate compares with a mortality rate of 50% in the other reports of penile gangrene as well as the 16 cases of diffuse calcinosis. Therefore, we recommend a conservative treatment approach with surgical intervention reserved for cases associated with severe infection or unremitting pain. Of our 7 patients 5 were treated with local wound care: 2 survived and 3 died of unrelated events. Furthermore, our experience suggests that elective circumcision should be avoided in patients with severe vascular disease associated with chronic renal failure and a high calcium-phosphorus product. Of our 7 patients 2 had distal penile gangrene as a result of circumcision and, thus, experienced a significant amount of morbidity due to the surgery. Patient 1, who had patchy gangrene on the glans, subsequently suffered complete gangrene of the distal shaft following circumcision necessitating total penectomy. Several investigators have concluded that parathyroidectomy may be beneficial. Despite parathyroidectomy, however, the mortality rate and need for subsequent limb amputation remained high in all reported series. As discussed previously, 1 patient who improved postoperatively had no hyperplasia on pathological examination. None of our patients underwent parathyroidectomy, with 71 % showing stabilization or improvement of the disease. This finding compares favorably with those of patients historically treated with parathyroidectomy, thus casting doubt on the efficacy of this treatment. CONCLUSIONS
Distal penile gangrene is a focal manifestation of diffuse arterial calcification found in patients with altered calcium metabolism as a result of chronic renal failure. The mortality rate of affected patients is high due to the severity of the associated illnesses. An aggressive surgical approach (excision of the gangrenous segment and/or subtotal parathyroidectomy) is not warranted, since the morbidity and mortality remain unchanged. Amputation of gangrenous areas is indicated only in patients with unremitting pain or when superinfection is not adequately controlled by antibiotics. Subtotal parathyroidectomy should be considered as a last resort, and then only in patients with diffuse, rapidly progressive calcinosis and severe secondary hyperparathyroidism that is
poorly controlled by medical therapy. Spontaneous resolution of patchy gangrene or necrotic tissue slough along lines of demarcation is common in untreated patients. Because patients with renal failure are living longer, prevention of this potential problem with a more aggressive medical approach to the altered calcium metabolism before the onset of calcinosis is indicated. REFERENCES
1. Ashouri, 0. S. and Perez, R. A.: Vascular calcification presenting as necrosis of penis in patient with chronic renal failure. Urology, 28: 420, 1986. 2. Lowe, F. C. and Brendler, C. B.: Penile gangrene: a complication of secondary hyperparathyroidism from chronic renal failure. J. Ural., 132: 1189, 1984. 3. Rosen, H., Friedman, S. A., Raizner, A. E. and Gerstmann, K.: Azotemic arteriopathy. Amer. Heart J., 84: 250, 1972. 4. Perloff, L. J., Spence, R. K., Grossman, R. A. and Barker, C. F.: Lethal post-transplantation calcinosis. Transplantation, 27: 21, 1979. 5. Gipstein, R. M., Coburn, J. W., Adams, D. A., Lee, D. B. N., Parsa, K. P., Sellers, A., Suki, W. N. and Massry, S. G.: Calciphylaxis in man. A syndrome of tissue necrosis and vascular calcification in 11 patients with chronic renal failure. Arch. Intern. Med., 136: 1273, 1976. 6. Hallgren, R., Wibell, L., Ejerblad, S., Eriksson, I., Johansson, H., Grimelius, L. and Wilander, E.: Arterial calcification and progressive peripheral gangrene after renal transplantation. Report of two cases treated with parathyroidectomy. Acta Med. Scand., 198: 331, 1975. 7. Conn, J., Jr., Krumlowsky, F. A., Del Greco, F. and Simon, N. M.: Calciphylaxis: etiology of progressive vascular calcification and gangrene. Ann. Surg., 177: 206, 1973. 8. Katz, A. I., Hampers, C. L. and Merrill, J.P.: Secondary hyperparathyroidism and renal osteodystrophy in chronic renal failure. Analysis of 195 patients, with observations on the effects of chronic dialysis, kidney transplantation and subtotal parathyroidectomy. Medicine, 48: 333, 1969. 9. Parfitt, A. M.: Soft-tissue calcification in uremia. Arch. Intern. Med., 124: 544, 1969. 10. Carroll, H.J. and Oh, M. S.: Disturbances in calcium, phosphate, and magnesium metabolism. In: Water, Electrolyte, and AcidBase Metabolism: Diagnosis and Management. Philadelphia: J. B. Lippincott Co., chapt. 8, pp. 306-355, 1978. 11. Chan, Y. L., Mahony, J. F., Turner, J. J. and Posen, S.: The vascular lesions associated with skin necrosis in renal disease. Brit. J. Derm., 109: 85, 1983.
THE JOURNAL OF UROLOGY Copyright© 1994 by AMERICAN UROLOGICAL AssOCIATION, INC.
PENILE GANGRENE ASSOCIATED WITH CHRONIC RENAL FAILURE: REPORT OF 7 CASES AND REVIEW OF THE LITERATURE MARK STEIN, CHRISTIAN ANDERSON, RICCARDO RICCIARDI, JOE W. CHAMBERLIN, SETH E. LERNER AND DANIEL GLICKLICH From the Departments of Urology and Medicine, Montefiore Medical Center, Albert Einstein College of Medicine, Bronx, New York
ABSTRACT
Distal penile gangrene associated with renal failure is a rare entity with only 3 cases reported in the literature. Certain physiological abnormalities are commonly found in association with this condition, including secondary hyperparathyroidism, diabetes and peripheral vascular disease. We report our experience with 7 patients who presented with this condition. All patients had end stage renal disease with 5 on hemodialysis, 1 on peritoneal dialysis and 1 with a functioning cadaveric renal transplant. Six patients had diabetes mellitus and all had derangements of the calcium and phosphate metabolism, with the calcium-phosphorus product being greater than 70. Five patients were treated expectantly with resolution of gangrene in 2 and stable disease in 3. Three of the 5 patients managed expectantly and both patients treated with penectomy died of unrelated causes within 3 months. We conclude that there is no advantage to aggressive surgical treatment of penile gangrene associated with renal failure, since the outcome is the same. The overall mortality for this group is high due to associated co-morbid disease regardless of the type of treatment. Furthermore, subtotal parathyroidectomy is not indicated, since spontaneous improvement and mortality rates reported in our series were similar to those of previously reported cases. Expectant management of affected patients affords the best treatment. KEY WORDS:
penis; gangrene; renal failure, chronic
Distal penile gangrene in patients with chronic renal failure is a rare entity, with only 3 cases in the literature. 1 - 3 Unlike Fournier's gangrene, this disease is limited to the glans penis or distal penile shaft and usually follows a relatively indolent course (see figure). Affected patients frequently suffer from disorders associated with renal failure, such as diabetes mellitus, hypertension and occlusive arterial disease. Distal penile gangrene is believed to be a variant of diffuse, rapidly progressive arterial calcification,4· 5 which is attributed to secondary hyperparathyroidism that occurs in patients with chronic renal failure, and leads to arterial occlusion and distal extremity loss. Historically, treatment has included surgical removal of the parathyroid and extirpation of gangrenous areas. A high associated mortality rate is reported despite this aggressive management. To define further this rare entity and improve upon the management strategy, we reviewed our experience with 7 patients who presented with distal penile gangrene and chronic renal failure during a 4-year period.
hyperparathyroidism consisting of an elevated calcium-phosphorus product. Patients 1 al).d 2 complained of worsening peripheral vascular disease. Patient 1 had rest pain in all 10 fingers as well as dry gangrene of the distal digits and patient 2 had severe left lower extremity ischemia requiring attempted balloon angioplasty of the left popliteal artery followed by left above the knee amputation. Patient 1 had focal gangrenous patches on the glans penis. Due to phimosis, which precluded local wound care, circumcision was performed. Gangrene progressed following circumcision and the patient underwent partial penectomy. The necrotic areas progressed despite local wound care and penectomy with perinea! urethrostomy
PATIENT INFORMATION
Between July 1989 and April 1993, 621 patients 50 to 87 years old (mean age 66.6 years) with chronic renal failure requiring dialysis were treated at our university medical center and 2 affiliated dialysis units. Of these patients urological consultation was requested for 7 to evaluate penile gangrene or penile pain. Detailed patient information is presented in table 1. Of the 7 patients 6 had diabetes mellitus. Six patients were dialysis dependent, while 1 had a functioning cadaveric renal allograft. All patients had clinical evidence of preexisting occlusive vascular disease, some with more than 1 type (5 with peripheral vascular disease, 4 with coronary artery disease and 1 with cerebrovascular disease). All patients had biochemical evidence of secondary Accepted for publication March 25, 1994.
Patient 7. Distal penile gangrene associated with renal failure 2014
2015
PENILE GANGRENE ASSOCIATED WITH CHRONIC RENAL FAILURE TABLE 1.
Pt.-Age No.
Age, dialysis status, medical history and calcium-phosphorus product in patients with penile gangrene Systemic Diseases
Dialysis (mos.)
1-71
Diabetes mellitus, end stage renal disease
Hemodialysis (12)
2-73
Diabetes mellitus, end stage renal disease, colon Ca Diabetes mellitus, myocardial infarction, congestive heart failure, end stage renal disease Intravenous drug abuse, chronic obstructive pulmonary disease, tuberculosis, end stage renal disease Diabetes mellitus, hypertension, congestive heart failure, end stage renal disease, cirrhosis Diabetes mellitus, cerebrovascular accident, myocardial infarction, end stage renal disease Coronary artery disease, diabetes mellitus, chronic obstructive pulmonary disease, end stage renal disease
Hemodialysis (5)
3-54 4-65 5-50 6-87 7-66
Transplant
Prior Vascular Disease
CalciumPhosphorus Product
Above knee amputation, pain and gangrene of digits Above knee amputation
71.5
Below knee amputation, rt. digit amputation, colostomy for ischemic bowel, coronary artery disease
75.9
Peritoneal dialysis (more than 24)
78.3
72.0
Hemodialysis (18)
Coronary artery disease
76.0
Hemodialysis (more than 24)
Lower extremity rest pain
77.3
Hemodialysis (more than 24)
Coronary artery disease
71.0
was later required. The patient died of overwhelming sepsis unrelated to the operation 3 months later. Patient 2 was managed expectantly and the gangrenous area appeared to stabilize. He died of a massive cerebrovascular accident soon after presentation. Two patients had distal penile gangrene following acute hypotensive episodes and myocardial infarction. Patient 3 had dry gangrene of the entire glans penis and was managed expectantly with autoamputation of the gangrenous segment within 4 weeks. Subsequently, cellulitis of the remaining area developed, which was treated with intravenous antibiotics and local wound care. The patient was discharged from the hospital and has had no further morbidity. Patient 4 had focal dry gangrenous patches on the glans penis. He was treated conservatively with topical antibiotic ointment with resolution of the gangrene. This patient died of overwhelming sepsis due to an unrelated cause 1 month later. Three patients presented with recurrent balanitis and phimosis, and were scheduled for elective circumcision. Patient 5 had gangrenous areas along the distal prepuce and glans before the elective circumcision. He was initially treated with circumcision and debridement of the necrotic areas. Subsequently, the devitalized areas progressed and partial penectomy was performed. He was discharged home but rehospitalized 2 months later with a polymicrobial necrotizing fasciitis of the leg and he died. Patients 6 and 7 underwent elective circumcision for phimosis. Subsequently, within 1 week gangrenous patches and total glandular gangrene developed, respectively. Both patients were managed conservatively with local wound care. Patient 6 improved and was discharged home without any further morbidity. The area of gangrene stabilized in patient 7 but he died of multi-organ system failure several months later. DISCUSSION
Diffuse vascular calcification is a manifestation of abnormal calcium and phosphorus metabolism that occurs in patients with chronic renal failure and in renal transplant recipients. 4 - 6 Arterial and metastatic soft tissue calcification occurs as a result of secondary hyperparathyroidism and its associated elevation of the serum calcium-phosphorus product. The risk for metastatic calcification is believed to be considerable when the calcium-phosphorus product exceeds 70. 7 - 9 The etiology of secondary hyperparathyroidism inuremic patients has been well described. 10 Histological examination of affected arteries reveals calcific infiltration of the media with reactive connective tissue hyperplasia of the in-
tima that results in luminal obliterations and ultimately progression to distal tissue gangrene. 3 • 11 There have been several reports in the literature of distal extremity gangrene associated with abnormal calcium metabolism. Gipstein et al reported on 11 patients with end stage renal disease, high or high normal serum calcium levels and severe hyperphosphatemia. 5 Of these patients 10 were treated with parathyroidectomy, of whom 7 had improvement in the ischemic lesions. They concluded that parathyroidectomy is an effective treatment and that early institution of phosphate binding agents may prevent this syndrome. Perloff et al reported on 4 patients with post-transplantation calcinosis, of whom 1 had distal penile gangrene. 4 All patients were treated with subtotal parathyroidectomy but only 1 survived to be discharged from the hospital. They recommended subtotal parathyroidectomy with autotransplantation despite the poor outcome and lack of correlation between the operation and healing of distal gangrenous ulcers. The penile gangrene in 1 patient in this series resulted from a spreading staphylococcal infection from the thigh, similar to Fournier's gangrene, and so this does not represent the type of penile gangrene discussed in our series. Fournier's gangrene is a form of bacterial necrotizing fasciitis that causes wet gangrene. Diffuse vascular calcification results in obliteration of arterial inflow, causing ischemic dry gangrene. Hallgren et al presented 2 cases of progressive arterial calcifications resulting in peripheral gangrene, treated by subtotal parathyroidectomy. 6 Of the 2 patients 1 improved postoperatively. In this case there was no evidence of parathyroid hyperplasia in the pathological specimen. One patient with mild glandular hyperplasia on pathological section died shortly following surgery. Isolated penile gangrene is believed to be a focal manifestation of diffuse arterial calcification seen in chronic renal failure patients. Lowe and Brendler described a patient who was initially managed conservatively until distal penectomy was required for control of ischemic pain. 2 Subtotal parathyroidectomy was performed to stabilize the vascular deterioration. The patient ultimately required amputation of 2 fingers for preexisting gangrene, and a left below the knee amputation for nonhealing foot and ankle ulcers. Ashouri and Perez reported on a case in which the necrotic areas were excised without further patient morbidity. 1 Our report expands the literature of penile gangrene as a result of diffuse arterial calcification associated with chronic renal failure from 3 cases to 10. The mortality rate for this
2016
PENILE GANGRENE ASSOCIATED WITH CHRONIC RENAL FAILURE TABLE
2. Presenting symptoms, clinical findings, treatment, outcome and mortality in patients with penile gangrene
Presenting Signs and Symptoms
Gross Description
Treatment
1
Worsening peripheral vascular disease
Focal gangrenous patches
Circumcision, partial and then total penectomy with perinea! urethrostomy
2
Entire glans
Observation
Entire glans
Observation, autoamputation, antibiotics, debridement Observation
Gangrene of penile tissues
4
Worsening peripheral vascular disease Shock after myocardial infarction Urosepsis, shock
5
Painful phimosis
Gangrene of distal glans, gram-pos. cocci
6
After circumcision
Circumcision, debridement, partial penectomy Observation
Pt. No.
3
Focal gangrenous patches Distal prepuce, glans Focal gangrenous patches Entire glans
Pathology Gangrene of glans and corpus cavernosum, thrombi, calcified small and medium vessels Gangrene of glans
Outcome Died of sepsis*
Died of myocardial infarction* Alive, serum creatinine 1.2 mg./dl.t Died of sepsist Died of sepsis* Alivet
Died of multi-organ Focal gangrene of foreskin system failuret All circumcisions were performed using the sleeve or dorsal slit technique, without the use of a tourniquet. Minimal, superficial cautery was used for hemostasis. * No progression of penile gangrene; died of unrelated causes within 3 months. t No further progression of penile gangrene. t Gangrenous area improved before death. 7
After circumcision
Observation
group remains high due to the severity of the associated systemic illnesses: 5 of the 7 patients died within 3 months after presentation (table 2). This rate compares with a mortality rate of 50% in the other reports of penile gangrene as well as the 16 cases of diffuse calcinosis. Therefore, we recommend a conservative treatment approach with surgical intervention reserved for cases associated with severe infection or unremitting pain. Of our 7 patients 5 were treated with local wound care: 2 survived and 3 died of unrelated events. Furthermore, our experience suggests that elective circumcision should be avoided in patients with severe vascular disease associated with chronic renal failure and a high calcium-phosphorus product. Of our 7 patients 2 had distal penile gangrene as a result of circumcision and, thus, experienced a significant amount of morbidity due to the surgery. Patient 1, who had patchy gangrene on the glans, subsequently suffered complete gangrene of the distal shaft following circumcision necessitating total penectomy. Several investigators have concluded that parathyroidectomy may be beneficial. Despite parathyroidectomy, however, the mortality rate and need for subsequent limb amputation remained high in all reported series. As discussed previously, 1 patient who improved postoperatively had no hyperplasia on pathological examination. None of our patients underwent parathyroidectomy, with 71 % showing stabilization or improvement of the disease. This finding compares favorably with those of patients historically treated with parathyroidectomy, thus casting doubt on the efficacy of this treatment. CONCLUSIONS
Distal penile gangrene is a focal manifestation of diffuse arterial calcification found in patients with altered calcium metabolism as a result of chronic renal failure. The mortality rate of affected patients is high due to the severity of the associated illnesses. An aggressive surgical approach (excision of the gangrenous segment and/or subtotal parathyroidectomy) is not warranted, since the morbidity and mortality remain unchanged. Amputation of gangrenous areas is indicated only in patients with unremitting pain or when superinfection is not adequately controlled by antibiotics. Subtotal parathyroidectomy should be considered as a last resort, and then only in patients with diffuse, rapidly progressive calcinosis and severe secondary hyperparathyroidism that is
poorly controlled by medical therapy. Spontaneous resolution of patchy gangrene or necrotic tissue slough along lines of demarcation is common in untreated patients. Because patients with renal failure are living longer, prevention of this potential problem with a more aggressive medical approach to the altered calcium metabolism before the onset of calcinosis is indicated. REFERENCES
1. Ashouri, 0. S. and Perez, R. A.: Vascular calcification presenting as necrosis of penis in patient with chronic renal failure. Urology, 28: 420, 1986. 2. Lowe, F. C. and Brendler, C. B.: Penile gangrene: a complication of secondary hyperparathyroidism from chronic renal failure. J. Ural., 132: 1189, 1984. 3. Rosen, H., Friedman, S. A., Raizner, A. E. and Gerstmann, K.: Azotemic arteriopathy. Amer. Heart J., 84: 250, 1972. 4. Perloff, L. J., Spence, R. K., Grossman, R. A. and Barker, C. F.: Lethal post-transplantation calcinosis. Transplantation, 27: 21, 1979. 5. Gipstein, R. M., Coburn, J. W., Adams, D. A., Lee, D. B. N., Parsa, K. P., Sellers, A., Suki, W. N. and Massry, S. G.: Calciphylaxis in man. A syndrome of tissue necrosis and vascular calcification in 11 patients with chronic renal failure. Arch. Intern. Med., 136: 1273, 1976. 6. Hallgren, R., Wibell, L., Ejerblad, S., Eriksson, I., Johansson, H., Grimelius, L. and Wilander, E.: Arterial calcification and progressive peripheral gangrene after renal transplantation. Report of two cases treated with parathyroidectomy. Acta Med. Scand., 198: 331, 1975. 7. Conn, J., Jr., Krumlowsky, F. A., Del Greco, F. and Simon, N. M.: Calciphylaxis: etiology of progressive vascular calcification and gangrene. Ann. Surg., 177: 206, 1973. 8. Katz, A. I., Hampers, C. L. and Merrill, J.P.: Secondary hyperparathyroidism and renal osteodystrophy in chronic renal failure. Analysis of 195 patients, with observations on the effects of chronic dialysis, kidney transplantation and subtotal parathyroidectomy. Medicine, 48: 333, 1969. 9. Parfitt, A. M.: Soft-tissue calcification in uremia. Arch. Intern. Med., 124: 544, 1969. 10. Carroll, H.J. and Oh, M. S.: Disturbances in calcium, phosphate, and magnesium metabolism. In: Water, Electrolyte, and AcidBase Metabolism: Diagnosis and Management. Philadelphia: J. B. Lippincott Co., chapt. 8, pp. 306-355, 1978. 11. Chan, Y. L., Mahony, J. F., Turner, J. J. and Posen, S.: The vascular lesions associated with skin necrosis in renal disease. Brit. J. Derm., 109: 85, 1983.