Successful Management of a Severe Sacral Pressure Injury Penetrating to the Retroperitoneum

Introduction. Reports of retroperitoneal infection related to a sacral pressure injury (PI) are rare, and none of the reports described the direct spread of infection through the sacrum to the retroperitoneum. The authors present, to their knowledge, the first report of a severely infected PI that showed full-thickness sacral destruction and direct retroperitoneal penetration. Case Report. A 63-year-old female was referred for management of a stage 4 sacral PI complicated by a retroperitoneal abscess. The patient’s comorbidities were diabetes mellitus and pemphigus foliaceus with steroid therapy-induced immunosuppression. Upon admission, the patient presented with a sacral PI producing copious purulent discharge that measured 5 cm × 3 cm. Magnetic resonance imaging revealed full-thickness sacral bone destruction and a massive retroperitoneal abscess, suggesting the sacral PI directly penetrated to the retroperitoneal space. Antibiotics were administered, and surgical debridement and sequestrectomy were performed. Negative pressure wound therapy (NPWT) with continuous saline irrigation was initiated. The patient’s mesorectum was exposed within the retroperitoneal space. Therefore, a nonadhesive wound dressing was applied before placing the irrigation tube to avoid perforating the rectum. Because the patient had fragile skin secondary to long-standing pemphigus foliaceus and steroid treatment, a liquid skin protectant and hydrocolloid wound dressing were applied. The infection was successfully controlled with NPWT with saline irrigation. The patient experienced no rectal injury or skin rupture, and surgical closure was performed after 75 days. Although partial wound dehiscence occurred because of the poor condition of the skin, the resultant open wound was managed conservatively. The patient showed no retroperitoneal abscess recurrence 6 months later. Conclusions. A rare case of an intractable sacral PI complicated by retroperitoneal abscess was successfully managed in an immunocompromised patient. Notably, NPWT with saline irrigation was useful in controlling the patient’s severe retroperitoneal infection.

Introduction

Pressure injuries (PIs) are common in aging societies.^1,2 Pressure injuries can become severe, especially in patients who are immunocompromised and those with poor nutritional status or multiple comorbidities. Pressure injuries considered to be late-stage require multidisciplinary treatments, including wound care, antibiotics, negative pressure wound therapy (NPWT), surgical debridement, and flap wound closure. However, these treatments are typically prolonged and require substantial effort from medical personnel, and they do not necessarily produce optimal outcomes.

Herein, the authors report, to their knowledge, the first case of a severely infected PI that showed full-thickness sacral destruction and direct retroperitoneal penetration. Although the patient’s injury was refractory to conventional treatments because of multiple comorbidities, including pemphigus foliaceus with steroid therapy-induced immunosuppression and diabetes mellitus, the PI was successfully managed with a multidisciplinary approach.

Case Report

A 63-year-old female with a 4-year history of an intractable sacral PI was transferred for management of increased purulent exudate from the wound. The patient had a 16-year history of pemphigus foliaceus, and the PI developed after the patient became bedridden secondary to worsening of pemphigus foliaceus in the legs. The patient took 1.5 g oral betamethasone daily, resulting in steroid therapy-induced diabetes mellitus. The patient also had hypertension, hyperlipidemia, osteoporosis, and low nutritional status (serum albumin level, 2.2 g/dL).

Upon admission, a stage 4 sacral PI³ with copious purulent discharge measuring 5 cm × 3 cm was noted (Figure 1A). Because of long-standing pemphigus foliaceus and steroid consumption, the skin was thin and easily damaged by normal contact. Wound cultures grew ampicillin/sulbactam-sensitive Streptococcus agalactiae, Lactobacillus sp, Corynebacterium sp, Bacteroides thetaiotaomicron, Prevotella buccalis, Finegoldia magna, Porphyromonas asaccharolytica, and anaerobic gram-negative bacilli. Magnetic resonance imaging revealed osteomyelitis with full-thickness bone destruction around the fifth sacral vertebra and a massive retroperitoneal abscess, suggesting the sacral PI had destroyed the distal sacrum and the infection directly penetrated to the retroperitoneal space (Figure 1B).

Intravenous ampicillin/sulbactam (3 g/day) was started and continued for 6 weeks to control the osteomyelitis. On day 3, wound debridement and sequestrectomy of the sacrum and coccyx was performed. A small bone defect was found at the base of the PI that connected directly to the largely exposed retroperitoneum (Figure 1C), which was filled with purulent discharge. After enlarging the bone defect by resecting the surrounding sequestrum, the mesorectum was found to be exposed in the retroperitoneal space (Figure 2A). On day 4, NPWT with continuous saline irrigation (50 mL/hour) using a continuous low-pressure suction unit (MERA Sacuum; Senko Medical Instrument Manufacturing Co.) was initiated.⁴ An irrigation tube was inserted through the enlarged bone defect directly into the retroperitoneum. A nonadhesive wound dressing (Mepilex Transfer; Mölnlycke Health Care) was placed on the mesorectum to avoid an injury from direct contact between the tip of the irrigation tube and surrounding organs (Figure 2B). Because the patient’s skin was fragile, a liquid skin protectant (Cavilon No Sting Barrier Film; 3M) was applied to the surrounding skin, and the wound edge was covered with a thin hydrocolloid dressing (Visiderm; Convatec); subsequently, negative pressure (-37.5 mm Hg) was applied (Figure 2C). The infection began to resolve, the exposed retroperitoneal space began to close, and healthy granulation tissue gradually covered the exposed mesorectum (Figures 3A–C). On day 36, the continuous saline irrigation was discontinued and NPWT (VAC Therapy; 3M) with negative pressure set at -75 mm Hg to -100 mm Hg was initiated.

On day 75, after confirming the infection was controlled, the wound was closed using a superior gluteal artery perforator flap. The flap initially had inadequate blood flow, and pemphigus foliaceus-related epidermal peeling complicated the skin closure. Postoperatively, some dermal sutures failed, necessitating further conservative wound management. On day 127, the sacral wound was almost closed, and the patient was transferred to another hospital. After 6 months, although a small PI remained, there was no recurrence of sacral osteomyelitis or the retroperitoneal abscess (Figure 3D).

Ethics

The ethical board of the authors’ hospital approved this report, and the patient provided written informed consent for its publication.

Discussion

In this case report, an advanced sacral PI complicated by a retroperitoneal abscess in a patient with pemphigus foliaceus was successfully treated using a multidisciplinary approach. The retroperitoneal abscess was controlled using NPWT with saline irrigation, and the exposed mesorectum was protected by using a nonadhesive dressing.

A patient with a PI has an estimated risk of mortality twice that of a patient without a PI.² Further, the mortality in patients with a PI complicated by a serious infection is as high as 57.1%.¹ As in the present case, high-grade PIs can lead to infection within the surrounding subdural, epidural, and intrapelvic spaces, and, in such cases, the mortality can be even higher.

Several factors are believed to have contributed to the development of a severe PI in this patient. Pemphigus foliaceus is characterized by the loss of cell-to-cell adhesion between the dermis and epidermis secondary to the formation of IgG autoantibodies against desmoglein.^5-7 The long history of pemphigus foliaceus with steroid therapy caused extreme skin fragility, and this was the primary cause of the intractable PI. Additionally, the patient’s uncontrolled steroid-induced diabetes mellitus contributed to delayed wound healing. Further, the patient’s bones were very brittle secondary to osteoporosis, leaving the patient vulnerable to full-thickness destruction of the fifth sacral vertebra. Together, these conditions contributed to this atypical PI.

To the authors' knowledge, only 3 cases of sacral PI-related retroperitoneal infection have been reported in the English literature to date. However, these cases did not demonstrate the direct spread of infection through the sacrum to the retroperitoneum.^8-10 The present case, to the authors' knowledge, is the first to show that a sacral PI can penetrate the sacral bone and cause a retroperitoneal abscess.

Typically, a retroperitoneal abscess occurs either spontaneously or as a postoperative complication after urological or colorectal surgery, and the mortality rate ranges from 1.5% to 50%.^1-15 Previously, surgical drainage was the mainstay of treatment, and, more recently, minimally invasive computed tomography (CT)-guided percutaneous drainage emerged as an alternative.¹¹ Although CT-guided percutaneous drainage is currently the preferred treatment, it is accompanied by relatively high rates of failure (> 10%) and recurrence (> 10%).^8,11

The patient’s PI and retroperitoneal abscess were treated by enlarging the sacral defect to completely remove the sequestrum and allow sufficient drainage of the retroperitoneal abscess and the introduction of NPWT with irrigation. Continuous saline irrigation contributed to early infection control, and negative pressure facilitated rapid retroperitoneal closure. Negative pressure wound therapy dressings are contraindicated for use in wounds that expose organs because of the risk of organ injury. Therefore, a nonadhesive wound dressing was placed on the mesorectum, allowing NPWT to safely continue and avoiding a rectal perforation.

This patient, to the authors' knowledge, was the first case in which a retroperitoneal abscess was treated by NPWT irrigation performed through a full-thickness sacral defect. Negative pressure wound therapy with continuous irrigation was considered superior to CT-guided drainage in controlling infection in the patient’s widely exposed retroperitoneal space and was believed to be the most appropriate treatment. Although NPWT is rarely indicated in patients with pemphigus foliaceus because of their skin fragility, meticulous skin care allowed a full course of NPWT in this patient.

This case demonstrates that invasive wound closure surgery might be unsuccessful in patients with fragile skin. The patient’s dermis was so fragile that sutures failed, resulting in partial wound dehiscence. Thus, wound closure should be cautiously applied in this population.

Despite this complication, the experience could facilitate treatment planning in patients with fragile skin and a severe PI associated with retroperitoneal abscess. Thorough surgical debridement and sequestrectomy, careful application of NPWT, and meticulous skin care can lead to successful treatment in these challenging cases.

Limitations

A major limitation of this single case report is that the findings may not draw a definitive conclusion. Future accumulation of data is needed to further solidify outcomes of this treatment.

Conclusions

In a patient who is immunocompromised, a sacral PI can cause full-thickness sacral bone destruction and directly penetrate to the retroperitoneal space. Even in such a situation, multidisciplinary treatments enabled successful management of an intractable PI complicated by a retroperitoneal abscess. Negative pressure wound therapy with saline irrigation can control a severe retroperitoneal infection related to a sacral PI.

Acknowledgments

Authors: Hideki Kadota, MD, PhD¹; Kayo Miyashita, MD¹; Seita Fukushima, MD¹; Chikafumi Oryoji, MD¹; Masuo Hanada, MD, PhD¹; Sei Yoshida, MD¹; Hayato Fujita, MD, PhD²; and Yukiko Tachibana, RN, WOCN³

Affiliations: ¹Department of Plastic and Reconstructive Surgery, Kyushu University Hospital, Japan; ²Department of Surgery and Oncology, Graduate School of Medical Sciences, Kyushu University, Japan; and ³Division of Pressure Ulcer Prevention, Graduate School of Medical Sciences, Kyushu University, Japan

Correspondence: Hideki Kadota, MD, PhD, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, Japan; kadohide@qent.med.kyushu-u.ac.jp, hkadota@hotmail.co.jp

Disclosure: Dr Kadota has received speaking fees from 3M+KCI. The remaining authors disclose no financial or other conflicts of interest.

References

1. Braga IA, Pirett CC, Ribas RM, Gontijo Filho PP, Diogo Filho A. Bacterial colonization of pressure ulcers: assessment of risk for bloodstream infection and impact on patient outcomes. J Hosp Infect. 2013;83(4):314–320. doi:10.1016/j.jhin.2012.11.008

2. Song YP, Shen HW, Cai JY, Zha ML, Chen HL. The relationship between pressure injury complication and mortality risk of older patients in follow-up: a systematic review and meta-analysis. Int Wound J. 2019;16(6):1533–1544. doi:10.1111/iwj.13243

3. Edsberg LE, Black JM, Goldberg M, McNichol L, Moore L, Sieggreen M. Revised National Pressure Ulcer Advisory Panel pressure injury staging system: revised pressure injury staging system. J Wound Ostomy Continence Nurs. 2016;43(6):585–597. doi:10.1097/WON.0000000000000281

4. Kiyokawa K, Takahashi N, Rikimaru H, Yamauchi T, Inoue Y. New continuous negative-pressure and irrigation treatment for infected wounds and intractable ulcers. Plast and Reconstr Surg. 2007;120(5):1257–1265. doi:10.1097/01.prs.0000279332.27374.69

5. Amagai M, Tanikawa A, Shimizu T, et al; Committee for Guidelines for the Management of Pemphigus Disease. Japanese guidelines for the management of pemphigus. J Dermatol. 2014;41(6):471–486. doi:10.1111/1346-8138.12486

6. Schmidt E, Kasperkiewicz M, Joly P. Pemphigus. Lancet. 2019;394(10201):882–894. doi:10.1016/S0140-6736(19)31778-7

7. Vinall C, Stevens L, McArdle P. Pemphigus vulgaris: a multidisciplinary approach to management. BMJ Case Rep. 2013;2013. doi:10.1136/bcr-2013-200479

8. Fujioka M, Yoshida S, Kitamura R, Matsuoka Y. Iliopsoas muscle abscess secondary to sacral pressure ulcer treated with computed tomography-guided aspiration and continuous irrigation: a case report. Ostomy Wound Manage. 2008;54(8):44–48.

9. Fukui K, Fujioka M, Ishiyama S. Sacral pressure ulcer-induced Fournier’s gangrene extending to the retroperitoneum: a case report. Wounds. 2018;30(1):E5–E8.

10. Yoon S, Jeong E, Lázaro HA. Multidisciplinary approach to an extended pressure sore at the lumbosacral area. Arch Plast Surg. 2016;43(6):586–589. doi:10.5999/aps.2016.43.6.586

11. Akhan O, Durmaz H, Balci S, Birgi E, Çiftçi T, Akinci D. Percutaneous drainage of retroperitoneal abscesses: variables for success, failure, and recurrence. Diagn Interv Radiol. 2020;26(2):124–130. doi:10.5152/dir.2019.19199

12. Capitán Manjón C, Tejido Sánchez A, Piedra Lara JD, et al. Retroperitoneal abscesses—analysis of a series of 66 cases. Scand J Urol Nephrol. 2003;37(2):139–144. doi:10.1080/00365590310008884

13. Coelho RF, Schneider-Monteiro ED, Mesquita JL, Mazzucchi E, Marmo Lucon A, Srougi M. Renal and perinephric abscesses: analysis of 65 consecutive cases. World J Surg. 2007;31(2):431–436. doi:10.1007/s00268-006-0162-x

14. Crepps JT, Welch JP, Orlando R 3rd. Management and outcome of retroperitoneal abscesses. Ann Surg. 1987;205(3):276–281. doi:10.1097/00000658-198703000-00010

15. Harris LF, Sparks JE. Retroperitoneal abscess. Case report and review of the literature. Dig Dis Sci. 1980;25(5):392–395. doi:10.1007/BF01308066

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