Giant haemophilic pseudotumour of the pelvis: case report and literature review

Pennekamp, P. H.; Strauß, Andreas C.; Klein, C. P. A. T.; Marx, Alexander; Goldmann, G.; Friedrich, M; Marquardt, Natascha; Oldenburg, Johannes

doi:10.1111/hae.12752

Cited by 7 publications

(5 citation statements)

References 6 publications

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“…Surgical removal is the primary treatment for large proximal pseudotumors or when conservative management fails 6 , 21 . Several case reports and retrospective analyses have shown that surgical resection is an effective and reasonable treatment for pseudotumors when the patient is covered by factor replacement therapy 19 , 22 , 23 , 24 , 25 . The cases described in this group belong to proximal pseudotumors, two cases appeared to have skin and bone destruction, and the operational indication was clear.…”

Section: Discussionmentioning

confidence: 99%

Outcome of Surgical Management of Hemophilic Pseudotumor: Review of 10 Cases from Single‐Center

Yao

Gao

et al. 2021

Orthopaedic Surgery

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Objective To evaluate the operative methods and clinical results of surgical treatment in a case series of 10 patients with hemophilic pseudotumors. Methods Ten patients with hemophilic pseudotumors who received surgical resection treatment in our hospital from October 2017 to June 2020 were retrospectively reviewed. All patients were hemophilia A (factor VIII deficiency).The age range was 20–51 years. Preoperative imaging examination revealed the size of irregular mass from 8.2 cm× 3.3 cm× 2.3 cm to 22.3 cm× 15.5 cm× 17.0 cm. With the supplementary of recombinant coagulation factor VIII, five cases received complete resection; one received resection and skin grafting; one received cytoreduction surgery as the pseudotumor closing to iliac vessel and nerve; three cases received complete resection and construction as bone destruction. The perioperative variables were recorded and all the patients were followed in the outpatient clinic. Clinical and radiological assessments were conducted. Results In these patients, the average intraoperative blood loss volume was 783.1 mL (range, 240–2100 mL). Six patients received blood transfusion during perioperative period. The average duration of surgery was 140.7 min (range, 110–240 min). All wounds healed smoothly and there was no infection or chronic sinus formation. The average length of hospital stay was 16.3 days (range, 12–25 days). There is no iatrogenic vascular nerve injury in our series. Complete follow‐up was performed in all patients. Mean follow‐up duration was 14.2 months (range, 6–26 months). One patient with pseudotumor in the thigh had a recurrence 1 year after operation, then secondary operation was performed. In three cases who received complete resection and construction, patient 8 obtained bone graft and late fixation. X‐ray examination showed bone formation in the lesion at the 2‐year follow‐ups after operation. Patient 9 underwent knee replacement, his left knee showed flexion deformity in preoparation. At the last follow‐up, range of motion was improved from 0° to 40° compared with preoperative status. Patient 10 had pseudotumor in the distal femur, received long bone graft and intramedullary nail fixation. Conclusions Surgical resection for hemophilic pseudotumors is an effective and safe method. The choice of surgical procedure must be individualized according to the localization and progress of pseudotumor.

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Section: Discussionmentioning

confidence: 99%

Outcome of Surgical Management of Hemophilic Pseudotumor: Review of 10 Cases from Single‐Center

Yao

Gao

et al. 2021

Orthopaedic Surgery

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“…Pennekamp et al [29] reported successful giant right iliac wing pseudotumour excision associated to a compound osteosynthesis with 20-hole reconstruction plate (attached to the remnants of the posterior superior iliac spine and the anterior acetabulum with screws and 3.0-mm), K-wires and bone cement to provide containment for the lesser pelvis and to fill dead space after resection of the destroyed ilium; the complication reported was the migration of a K-wire that required surgical revision because of the risk of rectal perforation.…”

Section: Discussionmentioning

confidence: 99%

Haemophilic Pelvic Pseudotumour: A New Surgical Option

et al. 2021

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Background: Haemophilia is an inherited coagulopathy caused by the absence or dysfunction of clotting factor VIII or IX. Clinical manifestations are generally secondary to recurrent bleeding episodes mainly in the musculoskeletal system. Bleeding symptoms appear early in life and, when the disease is severe (when plasma factor VIII or IX activity is <1% of normal), joint and muscle bleeding may occur spontaneously. A pseudotumour is a recurrent, chronic, encapsulated, slowly expanding, muscle hematoma. Haemophilic pseudotumour is a rare complication of haemophilia which occurs, as a condition either from repeated spontaneous bleeding or coming from a traumatic origin, in 1–2% of haemophilic patients. Case report: A 32-year-old man with severe haemophilia A referred to our Clinic with a massive right iliac wing pseudotumour complicated by Staphylococcus aureus superinfection and skin fistulisation. In this report we describe the medical management and surgical treatment by the adoption of a novel surgical technique which involves the use of a pedicle-screw and rod system (PSRS), a polyglycolic acid MESH and bone cement in order to build up an artificial ilium-like bony mass. This case report highlights the importance of interdisciplinary approach and the efficacy of eradicating surgery as treatment, especially in the case of large and long-lasting lesions.

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“…Surgical removal is the primary treatment for large proximal pseudotumours or when conservative management fails [11,14]. Several case reports and retrospective analyses have shown that surgical resection is an effective and reasonable treatment for pseudotumours when the patient is covered by factor replacement therapy [12,15,16,17,18]. Other therapeutic methods described in the literature include puncture and aspiration of the pseudotumour as well as aspiration and re lling of the pseudotumour with coralline hydroxyapatite [19.20].…”

Section: Discussionmentioning

confidence: 99%

Outcome of surgical management of hemophilic pseudotumor: Review of 9 Cases from single-center

Yao

Xue

Wang

et al. 2020

Preprint

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Background: Pseudotumours are a rare and serious complication of haemophilia, A slowly growing pseudotumour frequently destroys structures of bone and soft tissues. Surgical resection is the primary method for treatment of proximal pseudotumours. The purpose of this retrospective study was to evaluate the operative methods and clinical results of surgical treatment for those rare cases.Methods: We reviewed nine patients with hemophilic pseudotumours, who received surgical resection treatment in our hospital. The age range was 20-51 years. All operations were performed by one group surgeons. The method of operation depends on the origin and the extent of pseudotumor involvement. With the supplementary of recombinant coagulation factor VIII, six cases received complete resection; one received cytoreduction surgery as the pseudotumor closing to iliac vessel and nerve; two cases received complete resection and construction as bone destruction. Factor substitution was maintained for 14 days. Results: In our series, the average intraoperative blood loss volume was 710±35 ml(range ,240-2100ml). Six patients received blood transfusion during perioperative period. All wounds healed smoothly, no infection or chronic sinus formation. There is no iatrogenic vascular nerve injury in our series. Complete follow-up was performed in all patients. Mean follow-up duration was 14.5months (range, 6–26months). One patient with pseudotumor in the thigh had a recurrence of one year after operation, then secondary operation was performed, Conclusions: Surgical resection for haemophilic pseudotumours is an effctive and safe method . The choice of surgical procedure must be individualized according to the localization and the progress of pseudotumor. However, as relatively few cases and shorter follow-up time in our series. The long-term effects of these patients need further follow-up.

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Giant haemophilic pseudotumour of the pelvis: case report and literature review

Cited by 7 publications

References 6 publications

Outcome of Surgical Management of Hemophilic Pseudotumor: Review of 10 Cases from Single‐Center

Outcome of Surgical Management of Hemophilic Pseudotumor: Review of 10 Cases from Single‐Center

Haemophilic Pelvic Pseudotumour: A New Surgical Option

Outcome of surgical management of hemophilic pseudotumor: Review of 9 Cases from single-center

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