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Iowa Orthop J. 2010; 30: 174–176.
PMCID: PMC2958291

MARJOLIN'S ULCER: INCIDENTAL DIAGNOSIS OF SQUAMOUS CELL CARCINOMA ON HEMIPELVECTOMY FOR RECALCITRANT PELVIC OSTEOMYELITIS

Abstract

Hemipelvectomy is a radical surgical procedure reserved for particularly devastating pathology including recalcitrant pelvic osteomyelitis. We describe the incidental diagnosis of a metastatic squamous cell carcinoma by pathology after hemipelvectomy for pelvic osteomyelitis. This tumor was located deep within the chronic wound and deemed to be a Marjolin's ulcer (malignant transformation within a chronic wound). There are multiple reports and case series describing hemipelvectomy for tumor or infection, as well as one case report of a tumor arising years after successful surgical treatment of a chronic decubitus ulcer, but we were unable to find any describing the diagnosis of a Marjolin's ulcer at pathology following hemipelvectomy for osteomyelitis. This case demonstrates the diagnostic dilemma of malignant transformation within a chronic wound and is an opportunity to highlight the interventions necessary to prevent such progression.

CASE REPORT

A 66-year-old woman presented for treatment of a draining right decubitus ulcer in June of 2008. She had a history of T12 paraplegia following a motor vehicle accident in 1966. She was immunocompetent and without other contributing major comorbidities. She reported a 10 to 12 year history of ulcerations in this region with conservative management at her care facility. She was admitted to an outside hospital 4 weeks prior to her presentation for treatment of sepsis, presumably secondary to her decubitus wound. Physical exam demonstrated an 8 x 10cm foul-smelling wound in the right gluteal region that tracked down to ischium. She had two smaller and more superficial lesions in the left gluteal region. Examination of her lower extremities was also notable for ulcers of the bilateral heels. She had no motor or sensory function of her lower extremities bilaterally.

She was initially treated with surgical debridement of her right ischial wound including resection of the proximal femur and partial excision of the ischium. She also underwent a diverting colostomy. The excised ischial bone was sent for pathology and reported to be consistent with the diagnosis of osteomyelitis.

Wound cultures taken during the initial debridement grew Serratia marasacens and Prevotella. The Infectious Disease team was consulted during her first admission and all subsequent admissions. She was treated with broad spectrum antibiotics for an initial six week course. She was treated with negative pressure wound therapy as an outpatient. Her wound was healing well until June, 2009 at which time she presented with increasing drainage. She underwent irrigation and debridement with a rotational gluteus medius flap to her right decubitus ulcer. She presented again one month later with fevers and increasing drainage and was treated with repeat irrigation and debridement, followed by a similar presentation in September 2009 with another irrigation and debridement. She grew MRSA and Pseudomonas in cultures from these surgeries.

Given the recalcitrant nature of her pelvic osteomyelitis we discussed the possibility of hemipelvectomy. She elected to proceed with hemipelvectomy which was performed on 10/20/09 with the use of an anterior flap for coverage of her posterior ulcer. The procedure was complicated by a tear in the internal iliac vein during final amputation. This was repaired primarily. Two enlarged lymph nodes were encountered during the pelvic dissection near the bifurcation of the right internal and external iliac vessels. These nodes and the complete amputation specimen were sent for pathology.

The patient recovered remarkably well considering the magnitude of the operation. Her wound sealed in the first postoperative week. However, her pathology report determined she had a malignancy.

Pathology (6/2008):

Sections of bone show bony fragments and tra-beculae with marrow filled with fibrinopurulent debris. There are abundant neutrophils seen eroding into bony trabeculae.

Pathology (10/2009):

  1. Lymph node, pelvic, biopsy: Metastatic squamous cell carcinoma (1/1).
  2. Bone, pelvis, excision: Benign bone without significant inflammation or metastatic tumor.
  3. Leg and pelvis, right hindquarter amputation: Squa mous cell carcinoma, well differentiated, 14 cm, arising from a large ulcer in the posterior gluteal soft tissue with extension into underlying bone of pelvis and femur, anterior / medial soft tissue margin focally involved, bony margins not involved.
  4. Lymph node, site not specified, biopsy: Metastatic squamous cell carcinoma (1/1).

The diagnosis was discussed with the patient and the oncology team was consulted to discuss therapeutic options. A chest/abdomen/pelvis CT, ordered to stage the cancer, showed residual bilateral pelvic and inguinal lymphadenopathy as well as bilateral 1.2cm lung masses. A palliative approach was recommended by oncology. She was discharged 1 month following her hemipelvectomy but was subsequently readmitted 2 months postop-eratively for bowel obstruction. Exploratory laparotomy by general surgery revealed an incarcerated hernia of her colostomy with significant adhesions and metastatic disease. She had a prolonged intensive care unit stay for postoperative urosepsis. Attempts at weaning mechanical ventilation were unsuccessful. Her family chose to proceed with comfort measures only in accordance with her wishes and she passed away.

Figure 1
9/8/09 preoperative pelvis x-ray.
Figure 2
10/31/09 - postoperative CT.

DISCUSSION

Recalcitrant pelvic osteomyelitis is a potentially lethal disease that often requires aggressive measures to achieve a cure.1,2 The indications for hemipelvectomy in patients with pelvic osteomyelitis include sepsis and an intolerable state (i.e., foul-smelling wound intolerable to patient). In addition to the cardiopulmonary risks of any prolonged surgical intervention, hemipelvectomy carries with it the risks of intra-pelvic dissection including massive hemorrhage or bowel/bladder injury.1,2 The possibility of a Marjolin's ulcer should be considered prior to hemipelvectomy for recalcitrant pelvic osteomyelitis. The presence of such a tumor affects the predicted life expectancy of the patient as well as the goals of surgery.

The term Marjolin's ulcer was coined by Da Costa in his description of malignant degeneration of a chronic wound and referred to the initial description of this process by the French physician of the same name in 1828.3 These initial reports were both regarding tumors arising in vascular lesions without bony involvement and malignant transformation associated with osteomyelitis was not described until 1963.4 A recent literature review of Marjolin's ulcer reported that 76.5 percent of the cases included in the review occurred in burn scars, but that the types and locations of wounds varied greatly and included venous stasis ulcers, traumatic wounds, pressure sores, and osteomyelitis.5 Another recent review focused on Marjolin's ulcers specifically associated with osteomyelitis, quoting an incidence of cancerous transformation of 0.2% to 1.7% of chronic osteomyelitis cases.6 The vast majority of these tumors are squamous cell carcinomas, but fibrosarcoma, myeloma, lymphoma, plasmacytoma, angiosarcoma, rhabdomyosarcoma, and malignant fibrous histiocytoma have also been reported.7 Regardless of the nature of the wound in which they arise, these tumors are frequently aggressive and associated with metastasis at the time of diagnosis.8 The overall prognosis for patients with Marjolin's ulcer ranges from 65 to 75% for 3 year survival and this falls to 35 to 50% 3 year survival with metastasis.6,7,9-10 They are most likely to present after a chronic wound has been present for greater than 10 years and frequently for more than 40 years. Measures including proper positioning and skin care, early ulcer treatment, and frequent biopsy of chronic ulcers (> 10 years duration) remain the mainstays of preventing the long term sequalae of decubitus ulcers including both pelvic osteomyelitis and Marjolin's ulcer.11,12 Although there have been case reports of hemipelvectomy for eradication of Marjolin's ulcer, this surgical indication is rare given the aggressive nature of these tumors, likelihood of metastasis at diagnosis, and frequency of multiple comorbidities in these patients.13 Further highlighting the advantages of prevention over treatment, there has been a case report of Marjolin's ulcer developing at the site of a previously surgically treated and healed chronic decubitus ulcer.14

To our knowledge we have reported the first case of the incidental finding of squamous cell carcinoma on hemipelvectomy for pelvic osteomyelitis. It is likely that the recalcitrant nature of this patient's ulcer was secondary to the tumor. However, differentiating between recalcitrant osteomyelitis and Marjolin's ulcer is difficult given their similar and often simultaneous clinical presentations. The pathology results from the initial debridement confirmed pelvic osteomyelitis, at which time this tumor was likely present based on its size and metastasis at final pathology and the natural history of Marjolin's ulcer.

The clinical dilemma is not whether the diagnosis in such presentations is pelvic osteomyelitis or Marjolin's ulcer, but rather is the diagnosis pelvic osteomyelitis and Marjolin's ulcer. In hindsight, we recommend that excised soft tissue should be sent for pathology in addition to bone to evaluate for malignancy. A malignant transformation must always remain on the differential for an orthopedist treating a chronic, nonhealing wound, particularly in the setting of osteomyelitis.

REFERENCES

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10. Fleming MD, et al. Marjolin's ulcer: a review and reevaluation of a difficult problem. J Burn Care Rehabil. 1990;11:460–469. [PubMed]
11. Ellitsgaard N, et al. Development of carcinoma in chronic pressure sores in patients with spinal cord injuries [PubMed]
12. Eltorai IM, et al. Marjolin's ulcer in patients with spinal cord injury. J Spinal Cord Med. 2002;25(3):191–6. Fall. [PubMed]
13. Carlesimo B, et al. Hemipelvectomy and reconstruction in a patient with advanced Marjolin's ulcer: a case report. Int J Low Extrem Wounds. 2009;8(3):162–4. Sep. [PubMed]
14. Knudsen MA, Biering-Sorensen F. Development of Marjolin's ulcer following successful surgical treatment of chronic sacral pressure sore. Spinal Cord. 2008;46(3):239–40. Mar; Epub 2007 Jul 3. [PubMed]

Articles from The Iowa Orthopaedic Journal are provided here courtesy of The University of Iowa