Doxycycline sclerotherapy for pediatric head and neck macrocystic lymphatic malformations: A case series and review of the literature

Jamal, Nausheen; Ahmed, Sameer; Miller, Todd S.; Bent, John P.; Brook, Allan; Parikh, Sanjay R.; Ankola, Ashish A.

doi:10.1016/j.ijporl.2012.04.015

Cited by 42 publications

(25 citation statements)

References 18 publications

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“…Complications associated with treatment for LM around the airway have been reported in many papers. They include temporary conditions caused by sclerotherapy such as fever, local swelling pain, hemorrhage into the cyst, and infection . There also reported complications as the effects of treatment for head and neck lesions such as respiratory distress due to airway obstruction, as well as nerve palsy …”

Section: <Summary>mentioning

confidence: 99%

“…They include temporary conditions caused by sclerotherapy such as fever, 292,303,312,362,[365][366][367][368][369][370][371] local swelling 303,312,365,367,368,370,371 pain, 288,312,362,367,[370][371][372] hemorrhage into the cyst, 288,303,312,368 and infection. 288,292,303,310,312,360,365,372 There also reported complications as the effects of treatment for head and neck lesions such as respiratory distress due to airway obstruction, 292,303,306,312,362,365,366 as well as nerve palsy. 288,292,312,360,…”

Section: Complicationsmentioning

confidence: 99%

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Japanese clinical practice guidelines for vascular anomalies 2017

Mimura

Akita

Fujino

et al. 2020

Pediatrics International

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The objective was to prepare guidelines to perform the current optimum treatment by organizing effective and efficient treatments of hemangiomas and vascular malformations, confirming the safety, and systematizing treatment, employing evidence‐based medicine (EBM) techniques and aimed at improvement of the outcomes. Clinical questions (CQs) were decided based on the important clinical issues. For document retrieval, key words for literature searches were set for each CQ and literature published from 1980 to the end of September 2014 was searched in Pubmed, Cochrane Library, and Japana Centra Revuo Medicina (JCRM). The strengths of evidence and recommendations acquired by systematic reviews were determined following the Medical Information Network Distribution System (MINDS) technique. A total of 33 CQs were used to compile recommendations and the subjects included efficacy of resection, sclerotherapy/embolization, drug therapy, laser therapy, radiotherapy, and other conservative treatment, differences in appropriate treatment due to the location of lesions and among symptoms, appropriate timing of treatment and tests, and pathological diagnosis deciding the diagnosis. Thus, the Japanese Clinical Practice Guidelines for Vascular Anomalies 2017 have been prepared as the evidence‐based guidelines for the management of vascular anomalies.

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Section: <Summary>mentioning

confidence: 99%

Section: Complicationsmentioning

confidence: 99%

Japanese clinical practice guidelines for vascular anomalies 2017

Mimura

Akita

Fujino

et al. 2020

Pediatrics International

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“…19 Doxycycline's relative low cost, wide availability, and favorable safety profile have contributed to its common use as a sclerotherapy agent. 20,36 This study is the first to compare the outcomes of OK432 and doxycycline sclerotherapy for macrocystic and mixed LMs at a single institution by the same team of investigators.…”

Section: Introductionmentioning

confidence: 99%

OK432 Versus Doxycycline for Treatment of Macrocystic Lymphatic Malformations

Motz

Nickley

Bedwell

et al. 2014

Ann Otol Rhinol Laryngol

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Objectives: A variety of sclerotherapy agents are used to treat macrocystic lymphatic malformations (LMs). This retrospective study at a single institution was performed to compare the outcomes of pediatric macrocystic LMs of the head and neck that were treated with doxycycline or with OK432. Methods:The outcomes measured included early response to therapy, number of treatments required, operating room time, and adverse events. Results:The rates of clinical success for OK432 and doxycycline were similar (83% and 82%, respectively; p > 0.05), although OK432-treated patients required more treatments than did doxycycline-treated patients (1.9 versus 1.0 injections; p = 0.01; 95% confidence interval, 1.57 to 0.27). The average operating room time for a single OK432 injection was significantly shorter than that for doxycycline (53.2 versus 98.1 minutes; p < 0.001); however, when the total number of treatments administered was considered, the overall times in the operating room were similar. Adverse events in the early postoperative period were more common in OK432-treated patients, who experienced marked postoperative swelling compared to doxycycline-treated patients.Conclusions: OK432 and doxycycline are both effective sclerosants for the treatment of predominantly macrocystic LMs. The administration time for OK432 is shorter than that for doxycycline, but OK432 required more treatments overall to achieve clinical success. Early adverse events were more common in OK432-treated patients, but longer follow-up is necessary to determine whether rates of recurrence and adverse events are similar, particularly in light of the risk of tooth discoloration in doxycycline-treated patients.

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“…Doxycycline is often the primary sclerosant used in lymphatic malformations 22 and has been gaining popularity as a sclerosant because of its low cost, availability, and safety profile. 22,23 In cases of lymphatic malformations, success rates of 67% to 100% have been reported with doxycycline. 11,22,23 Doxycycline sclerotherapy has also been used successfully in Morel-Lavallée lesions, pleural effusions, and mixed capillary and lymphatic malformations.…”

Section: Discussionmentioning

confidence: 99%

“…22,23 In cases of lymphatic malformations, success rates of 67% to 100% have been reported with doxycycline. 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.…”

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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Doxycycline sclerotherapy for pediatric head and neck macrocystic lymphatic malformations: A case series and review of the literature

Cited by 42 publications

References 18 publications

Japanese clinical practice guidelines for vascular anomalies 2017

Japanese clinical practice guidelines for vascular anomalies 2017

OK432 Versus Doxycycline for Treatment of Macrocystic Lymphatic Malformations

Intramuscular Vascular Malformations

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