International Journal of Diabetes Mellitus 2 (2010) 196198

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International Journal of Diabetes Mellitus

journal homepage: www.elsevier.com/locate/ijdm

Case Report

Spontaneous gas gangrene of the scrotum in patient with severe diabetic ketoacidosis
Chu Zhang, Lizhen Ma, Fengying Peng, Yin Wu, Yu Chen, Yuhong Zhan, Xianfeng Zhang
Department of Endocrinology and Metabolism, Hangzhou Hospital, Nanjing Medical University, China

a r t i c l e

i n f o

a b s t r a c t
A 52-year-old Chinese man presented with severe diabetic ketoacidosis and markedly inated scrotum. Computed tomographic scans of the lower pelvis show extensive gas accumulation and inammatory changes in both sides of the scrotum. Gas gangrene of the scrotum was diagnosed and radical debridement along with other proactive anti-ketoacidosis therapy was performed immediately. Clostridium perfringens was found in cultures of necrotic tissue that veried the diagnosis and the patient was cured through multiple proactive treatments. 2010 International Journal of Diabetes Mellitus. Published by Elsevier Ltd. All rights reserved.

Article history: Received 26 September 2010 Accepted 30 September 2010

Keywords: Diabetic ketoacidosis Ketosis-prone T2DM Fourniers gangrene Clostridium perfringens

1. Introduction Spontaneous gas gangrene of the scrotum, also known as Fourniers gangrene (FG) is an extremely rare but life-threatening skin and soft tissue infective disease in the perineal region [1]. The infection commonly starts as cellulitis adjacent to the portal of entry, and rapidly progresses to extensive tissue necrosis. Without aggressive treatment, the patient will die from sepsis and multiple organ failure [2]. Multiple microbial infections by aerobes and anaerobes are always found in cultures from the wounds, most of which are normal commensals in the perineum and genitalia. Because of the impaired host cellular immunity, these conditional pathogens become virulent, and act synergistically to invade tissue and cause extensive damage [3]. Some comorbid systemic disorders with cellular immunity impairment, including AIDS, diabetes, alcohol abuse, leukemia, chemotherapy and chronic corticosteroid use [4] are identied in patients with FG. Diabetic ketoacidosis (DKA) is an acute complication of diabetes, characterized by circulation failure and acidosis. In developing countries, even with improved healthcare systems and reliable insulin supply, mortality and morbidity from DKA remain high

[5]. Although impaired tissue perfusion and defective immune response presented in DKA could be predisposing conditions for FG, there has been no previous report of specically pinpointing the DKA with FG. In the present case, we show how a patient with severe DKA and FG recovered through prompt, accurate diagnosis and emergent intervention.

2. Case presentation A 52-year-old previously healthy man was hospitalized on account of poor health. He mainly complained about polydipsia, fever and shortness of breath for 9 days, and a swollen scrotum was found 4 days later. Fever (always) occurred after rigors, and his maximum temperature reached 38.8 C. The swollen scrotum was accompanied by urodynia, hematuria and dysuria. He had not previously been diagnosed as diabetic, but his father had had type 2 diabetes (T2DM) for 34 years. Upon admission, he was in confusion, with a body temperature of 38.6 C, systolic blood pressure at 145 mm Hg, pulse at 96/min and breath rate at 24/min. His height was 174 cm, his body weight 82 kg and BMI 27. His skin was dry, and his extremities were cold. The scrotum was found to be swollen almost as big as a football, and was of a dark red colour; there was no tenderness but crepitation could be heard on touching. Laboratory examination revealed DKA and leucocytosis; plasma sugar was 16.95 mmol/L, blood WBCs was 38.2 109/L, plasma beta-hydroxybutyrate was 1898 mmol/L, urine acetone was 3+, urine lencocyte count was 20 per high power lens, pH was 7.06, HCO3 was 4 mmol/L, BE was 28 mmol/L, GAD-Ab, ICA and IAA

Abbreviations: DKA, diabetic ketoacidosis; WBCs, white blood cells; DM, diabetes mellitus; LADA, late onset autoimmune diabetes in adult; MRI, magnetic resonance imaging; CT, computed tomography. Corresponding author. Address: Department of Endocrinology and Metabolism, Hangzhou Hospital, Nanjing Medical University, Xueshi Road 4[#], Hangzhou City, Zhejiang Province 310006, China. Tel.: +86 0571 87065701. E-mail address: zhangxianfeng@medmail.com.cn (X. Zhang).

1877-5934/$ - see front matter 2010 International Journal of Diabetes Mellitus. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ijdm.2010.09.004

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were negative. Ultrasonography showed a lled cyst with hydronephrosis in both sides. A Lower Pelvis CT scan revealed extensive gas accumulation within the scrotum and inammatory changes to the scrotum wall (Fig. 1). DKA and spontaneous gas gangrene of the scrotum was diagnosed, conventional DKA treatment with uid replacement and continual insulin infusion were given, 6000 ml isotonic saline water was given in rst 24 h, thorough debridement and drainage were performed immediately, the wound surface was thoroughly rinsed with hydrogen peroxide, Tazocin and clindamycin were given as empirical antibiotic therapy. Cultures of necrotic tissue and urine from a urethral catheter showed Clostridium perfringens, and an antibiotic sensitivity test justied the empirical antibiotic choice. The antibiotic sensitivity test also showed that the patient was piperacillin, cefoperazone, clindamycin, Doxycycline, ooxacin, and metronidazole sensitive. Patient recovered consciousness in rst day of admission, temperature dropped back to normal range on the 4th day. He was given tazocin and clindamycin for 4 weeks. When he was discharged from the hospital 4 weeks later, his blood glucose was well controlled through insulin, and the infected scrotum was completely healed. When the patient went back clinic for a routine visit 6 months later, we discontinued his insulin therapy and solely used metformin; his glucose was well controlled thereafter. 3. Discussion FG is a rare, fulminant form of infective necrotizing fasciitis of the perineal, genital, or perianal regions, characterized by rapid progression of necrotising inammation and by the violent life-threatening course of the disease. Without appropriate treatment, the patient soon dies after septic shock and multiorgan failure [2]. DM is reported to be present in 2070% of patients with Fourniers gangrene [6], and the mortality rate of Fourniers gangrene is higher in patients with DM [7], as increased tissue glucose, poor tissue perfusion, and weak immune response in DM lead to a faster progress of infection and rapid development of systemic sepsis. DKA is an acute complication of DM, characterized by haemodynamic instability and acidosis, pathophysiological states facilitating FG to occur, and also being promoted by FG. Although such a severe infection like FG could be one factor that causes the initiation and development of DKA, these are often seen in patients with type 1 DM. The patient in the present case was in his fties, with

negative GAD-Ab, ICA and IAA, and his BMI was 27. All these are inconsistent with the diagnosis of type 1 DM or LADA. As severe DKA developed in such a short time, combined with other characteristics of patient, such as the age, BMI and negative autoantibody, we proposed that the patient be diagnosed as ketosis-prone T2DM. Another characteristic of ketosis-prone T2DM is that insulin secretion is markedly impaired at presentation, but intensied diabetic management results in signicant improvement in beta-cell function and insulin sensitivity, sufcient to allow discontinuation of insulin therapy within a few months of follow-up [8]. Actually, the patient in the present case switched his therapy from insulin to oral anti-diabetic glucose 6 months later, and this also supported the diagnosis of ketosis-prone type 2 diabetes. FG commonly starts as cellulites adjacent to the portal of entry. For those idiopathic cases, the source of infection can be ascribed to the gastrointestinal and genitourinary tract [9]. In the present case, without evidence of cutaneous injury or perianal abscess, most possibly, it was the infection of the urinary tract that initiated gas gangrene in the scrotum, as urine leucocyte counts was 20 per high power lens and culture of urine from urethral catheter showed Clostridium perfringens, the same pathogen in necrotic tissue. Clostridium perfringens, the organism responsible for classical myonecrotic gas gangrene, is frequently identied along with other bacteria in FG [10]. Under normal conditions, Clostridium species colonize in gastrointestinal and genitourinary tract, and become pathogens when predisposing factors are present, such as compromised immunity, diabetes, or invasive procedures within the urinary tract, gastrointestinal tract, or skin. In the local wounds of patients with FG, they produce various exotoxin and enzymes such as a toxin, collagenase, heparinase, and hyaluronidase, which lead to tissue necrosis, gas accumulation and the spread of infection[11]. Once FG is diagnosed, aggressive debridement of local wounds is warranted, along with other medical intervention like uid replacement for haemodynamic stabilization, and multi broad spectrum antibiotics, to cover all possible organisms. For patients of DKA and FG, the signicance of uid replacement is even more vital than it is for patient with DKA or FG alone, since both DKA and FG are in favor of circulation failure. The usual antibiotic combination includes penicillin or clindamycin for the streptococcal species, third generation cephalosporin or carbapenems for the gram negative organisms, plus metronidazole for the anaerobes [12]. Hyperbaric oxygen could be used as an adjunctive therapy

Fig. 1. (A) sagittal, (B) coronal, and (C) axial CT scan shows an accumulation of gas in the scrotum (white arrow) and extensive inammation of scrotum wall.

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C. Zhang et al. / International Journal of Diabetes Mellitus 2 (2010) 196198 [2] Pawlowski W, Wronski M, Krasnodebski IW. Fourniers gangrene. Pol Merkuriusz Lek 2004;17:857. [3] Rotstein OD, Pruett TL, Simmons RL. Mechanisms of microbial synergy in polymicrobial surgical infections. Rev Infect Dis 1985;7:15170. [4] Anaya DA, Dellinger EP. Necrotizing soft-tissue infection: diagnosis and management. Clin Infect Dis 2007;44:70510. [5] Otieno CF, Kayima JK, Omonge EO, Oyoo GO. Diabetic ketoacidosis: risk factors, mechanisms and management strategies in sub-Saharan Africa: a review. East Afr Med J 2005;82:S197203. [6] Morpurgo E, Galandiuk S. Fourniers gangrene. Surg Clin North Am 2002;82:121324. [7] Korkut M, Icoz G, Dayangac M, Akgun E, Yeniay L, Erdogan O, et al. Outcome analysis in patients with Fourniers gangrene: report of 45 cases. Dis Colon Rectum 2003;46:64952. [8] Umpierrez GE, Smiley D, Kitabchi AE. Narrative review: ketosis-prone type 2 diabetes mellitus. Ann Intern Med 2006;144:3507. [9] Asci R, Sarikaya S, Buyukalpelli R, Yilmaz AF, Yildiz S. Fourniers gangrene: risk assessment and enzymatic debridement with lyophilized collagenase application. Eur Urol 1998;34:4118. [10] Stevens DL. The pathogenesis of clostridial myonecrosis. Int J Med Microbiol 2000;290:497502. [11] Rood JI. Virulence genes of Clostridium perfringens. Annu Rev Microbiol 1998;52:33360. [12] Kobayashi S. Fourniers gangrene. Am J Surg 2008;195:2578.

in the treatment of FG, even though there is no conclusive evidence regarding its effectiveness. In conclusion, DKA with FG is a rare clinical emergency, but mortality on account of the disease is high, and early diagnosis and proactive medical intervention remain critical for patients survival. Clinical presentation in many patients during early stage may not be prominent. The detection of gas in the tissue by X-ray, CT, MRI or Ultrasonography should be a warning sign pointing to spontaneous gas gangrene. On the other hand, the proactive management of perineal injury or infection of patients with DKA or diabetes is also of extreme importance, in case FG may develop in these situations. Conict of interest None. Reference
[1] Smith GL, Bunker CB, Dinneen MD. Fourniers gangrene. Br J Urol 1998;81:34755.