Mixed Capillary/Lymphatic Malformation with Coexisting Port-Wine Stain: Treatment Utilizing 3D MRI and CT-Guided Sclerotherapy

Wimmershoff, M. B.; Schreyer, A. G.; Glaessl, Alexander; Geißler, A.; Hohenleutner, Ulrích; Feuerbach, Stefan; Landthaler, Míchael

doi:10.1046/j.1524-4725.2000.00004.x

Cited by 12 publications

(3 citation statements)

References 25 publications

(39 reference statements)

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“…11,22,23 Doxycycline sclerotherapy has also been used successfully in Morel-Lavallée lesions, pleural effusions, and mixed capillary and lymphatic malformations. 4,28,33,37 The exact mechanism of action remains unclear, but in animal pleurodesis models, doxycycline induces an inflammatory reaction resulting in fibrin and collagen deposition. 21 There is also evidence that doxycycline inhibits matrix metalloproteinase and suppresses vascular endothelial growth factor, which promotes angiogenesis.…”

Section: Discussionmentioning

confidence: 99%

Intramuscular Vascular Malformations

Mautner

Sussman

2014

Am J Sports Med

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Intramuscular vascular malformations are a rare subset of vascular lesions and present a considerable diagnostic challenge. The majority of vascular lesions are initially misdiagnosed 17,20,31 and are often attributed to more common musculoskeletal disorders. 14,30,38 Sports medicine practitioners need to be familiar with intramuscular vascular malformations because up to 20% of cases have symptoms of exertional leg pain. 20 Exertional leg pain, a common symptom seen by sports medicine practitioners, is defined as exercise-related leg pain distal to the knee and proximal to the talocrural joint. Depending on the study methodology, exertional leg pain has an incidence of 12.8% to 82.4% in athletes. 29 Although a comprehensive review of exertional leg pain is beyond the scope of this report, the differential diagnosis is broad and includes musculoskeletal, vascular, and neurologic etiologies (Table 1).Here we present a case of intramuscular venous malformation in the flexor digitorum longus muscle in a 15-yearold female runner with exertional leg pain. Treatment options for intramuscular venous malformations include observation, sclerotherapy, or surgical resection. Although a variety of different sclerosing agents have been described in the literature, to our knowledge, this is the first report of doxycycline sclerotherapy for intramuscular venous malformations. CASE REPORTA 15-year-old female runner presented with a 1-year history of exertional leg pain that gradually began while training for cross-country running. There was no associated trauma. Since initially developing symptoms, the patient had proceeded to participate in cross-country running, basketball, and lacrosse at her high school.Initially the pain was present only during running, but it progressed so that the patient started experiencing pain when walking and then eventually at rest. The pain localized to the left anteromedial lower leg. The patient reported associated left lower extremity swelling, occasional numbness and tingling in all 5 toes, and intermittent nondisabling low back pain. Her medical history was not significant.When symptoms first occurred 1 year prior, the patient initially saw an orthopaedic surgeon. At the time, a radiograph of the left lower extremity was obtained, with normal results. The patient was treated for a presumptive tibial stress fracture with a walking boot, but after 6 weeks of nonoperative management, the patient remained symptomatic. She was started on diclofenac, with minimal relief, and was prescribed physical therapy. Attempts at stretching, strengthening, and soft tissue manipulation worsened the patient's symptoms, and therapy was discontinued after only a few sessions.Further workup, including magnetic resonance imaging (MRI) of the lumbar spine and MRI of the left lower extremity without contrast, demonstrated normal results. Compartment pressure testing was performed with a handheld compartment pressure-testing device (Stryker Corporation) and was normal (before exercise: anterior = 11 mm Hg, lateral = 13...

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Section: Discussionmentioning

confidence: 99%

Intramuscular Vascular Malformations

Mautner

Sussman

2014

Am J Sports Med

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“…[ 4 ] Although rare, there have been a few reported cases of combined capillary–lymphatic malformation in individuals with PWS. [ 5 ] However, most of these cases were associated with syndromes such as Proteus syndrome and Klippel–Trenaunay syndrome.…”

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confidence: 99%

Port-Wine Stain with Rapid Hypertrophy and Nodularity: A Rare Case of Combined Capillary–Lymphatic–Arteriovenous Malformation

Dev,

Khunger,

Rishi

et al. 2024

Indian Journal of Dermatology

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“…The classical Hamburg classification of arterial, venous, lymphatic, arteriovenous, and combined vascular malformations can be further differentiated into truncal and extratruncal malformations. Truncal malformations are caused by a dysembryogenesis of the mature blood vessels, whereas extratruncal malformation derives from the primitive capillary network (1), as well as from both the lymphatic system of the skin and subcutaneous tissue (2).…”

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confidence: 99%

Preliminary communication: imiquimod in mixed capillary/lymphatic malformation

Bongiorno

Aricò

2008

Dermatologic Therapy

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The present authors reported a 14-year-old white boy who visited the present authors' dermatology department in January 2004. Physical examination revealed multiple translucent and hemorrhagic vesicles and skin-colored nodules on the chin. The lesion had grown slowly in size over the previous 7 years. The objective of this study is to estimate the exact mechanism of action of topical imiquimod on mixed capillary/lymphatic malformation. After 4 weeks of therapy the lesions were less protuberant. At the follow-up examination after a further 2 months of therapy, there was partial clinical regression of the capillary component with a return to normal skin color. One month after termination of therapy the lesions had completely regressed and there was no evidence of recurrence of the hemangiomatous section. The present authors' case suggests the efficacy of the use of topical imiquimod and this therapeutic modality may be of particular benefit in superficial type of capillary/lymphatic malformation, in which the destructive intervention may be undesirable.

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Mixed Capillary/Lymphatic Malformation with Coexisting Port-Wine Stain: Treatment Utilizing 3D MRI and CT-Guided Sclerotherapy

Abstract: The combination of CO2 laser treatment and sclerotherapy with doxycycline seems to present a treatment option for cutaneous and subcutaneous lymphangioma circumscriptum with rare side effects.

Cited by 12 publications

References 25 publications

Intramuscular Vascular Malformations

Intramuscular Vascular Malformations

Port-Wine Stain with Rapid Hypertrophy and Nodularity: A Rare Case of Combined Capillary–Lymphatic–Arteriovenous Malformation

Preliminary communication: imiquimod in mixed capillary/lymphatic malformation

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