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• W2093586341 abstract "Cutaneous leiomyoma is a rare benign tumor that is derived from the arrector pili muscle. Females with multiple cutaneous leiomyomas may also have uterine leiomyomas. This autosomal dominant genetic disorder is called Reed syndrome or familial leiomyomatosis cutis et uteri and is caused by a gene mutation located on chromosome 1q42.3-Q43.1Holst V.A. Junkins-Hopkins J.M. Elenitsas R. Cutaneous smooth muscle neoplasms: clinical features, histologic findings, and treatment options.J Am Acad Dermatol. 2002; 46: 477-490Abstract Full Text Full Text PDF PubMed Scopus (199) Google Scholar, 2Alam N.A. Bevan S. Churchman M. et al.Localization of a gene (MCUL1) for multiple cutaneous leiomyomata and uterine fibroids to chromosome 1q42.3-Q43.Am J Hum Genet. 2001; 68: 1264-1269Abstract Full Text Full Text PDF PubMed Scopus (120) Google Scholar Patients with cutaneous leiomyomata suffer from pain attacks, precipitated by cold or pressure, which can be severe.1Holst V.A. Junkins-Hopkins J.M. Elenitsas R. Cutaneous smooth muscle neoplasms: clinical features, histologic findings, and treatment options.J Am Acad Dermatol. 2002; 46: 477-490Abstract Full Text Full Text PDF PubMed Scopus (199) Google Scholar Several medications have been used with varying success to ameliorate the pain of multiple leiomyomata: phenoxybenzamine, nifedipine, oral nitroglycerin, beta-blockers, topical hyoscine hydrobromide, and gabapentin.3Alam M. Rabinowitz A.D. Engler D.E. Gabapentin treatment of multiple piloleiomyoma-related pain.J Am Acad Dermatol. 2002; 46: S27-S29Abstract Full Text Full Text PDF PubMed Scopus (61) Google Scholar, 4Archer C.B. Whittaker S. Greaves M.W. Pharmacological modulation of cold-induced pain in cutaneous leiomyomata.Br J Dermatol. 1988; 118: 255-260Crossref PubMed Scopus (44) Google Scholar, 5Batchelor R.J. Lyon C.C. Highet A.S. Successful treatment of pain in two patients with cutaneous leiomyomata with the oral alpha-1 adrenoceptor antagonist, doxazosin.Br J Dermatol. 2004; 150: 770-795Crossref PubMed Scopus (30) Google Scholar We report a patient with multiple painful cutaneous leiomyomata and uterine leiomyomas after emergency laparotomy for intra-abdominal bleeding, in whom relief from pain was achieved with pregabalin and duloxetine. A 34-year-old woman, American Society of Anesthesiologists Physical Status II, was admitted to our hospital due to a recurrent (habitual) abortion. Regarding her medical history, the patient had been operated on twice in the past for uterine fibroids and she also underwent diagnostic hysteroscopy. She reported onset of a brown painful node on her left shoulder at the age of 12 years. Over the years, further lesions developed on the chest and on the abdomen, which afterward became painful. The pain was spontaneous, having stabbing, burning, and pinching qualities, with numbness, and was exacerbated with exposure to cold, pressure, and emotional stress. During the years of worsening pain, although a variety of treatments had been received, including electrocoagulation, laser, physiotherapy, and nonsteroidal anti-inflammatory drugs, the pain was never satisfactorily controlled. Finally, the patient refused continuing treatment with either nifedipine or amitriptyline due to side effects. The patient was referred to the pain treatment unit of our department. Pain relief was achieved with the combined therapy of venlafaxine 75 mg daily, gradually increased to 600 mg daily (300 mg twice daily) and pregabalin 150 mg daily, progressively increased to 600 mg daily (300 mg twice daily). The increase of venlafaxine to a maximal dose of 600 mg/daily was decided by our psychiatrist to treat the patient's depression. Using a numeric pain intensity scale (NRS), pain was reduced from 10 to 1 (NRS range: 0=no pain to 10=unbearable pain). With this, a burning sensation was moderately improved after six months (NRS range: 10 to 5). In spite of this, symptoms recurred eight months later, when the patient decided to discontinue the medical therapy. She was commenced again on pregabalin 150 mg daily, gradually increased to 600 mg daily (300 mg twice daily) and duloxetine 60 mg daily instead of venlafaxine, which was not well tolerated. Interestingly, the patient reported complete relief of pain, even regarding the burning sensation. Now, 12 months later, the patient is still under combined treatment with pregabalin and duloxetine. She describes only short pain attacks, one or two monthly (NRS: 2–3). However, she regards those episodes of pain as controllable. Although the pathogenesis of pain that accompanies these cutaneous lesions is not known, it is suggested that pain is the result of the local pressure on the cutaneous nerves by the tumor. Muscle contraction mediated by alpha adrenergic receptors and/or the presence of a specific type of infiltrating cells represent possible mechanisms that may play a crucial role in pain induction.5Batchelor R.J. Lyon C.C. Highet A.S. Successful treatment of pain in two patients with cutaneous leiomyomata with the oral alpha-1 adrenoceptor antagonist, doxazosin.Br J Dermatol. 2004; 150: 770-795Crossref PubMed Scopus (30) Google Scholar A number of medications have been used for the treatment of pain of multiple leiomyomata with varying success, such as nifedipine, phenoxybenzamine, beta-blockers, oral nitroglycerin, topical hyoscine hydrobromide, and gabapentin.3Alam M. Rabinowitz A.D. Engler D.E. Gabapentin treatment of multiple piloleiomyoma-related pain.J Am Acad Dermatol. 2002; 46: S27-S29Abstract Full Text Full Text PDF PubMed Scopus (61) Google Scholar, 4Archer C.B. Whittaker S. Greaves M.W. Pharmacological modulation of cold-induced pain in cutaneous leiomyomata.Br J Dermatol. 1988; 118: 255-260Crossref PubMed Scopus (44) Google Scholar, 5Batchelor R.J. Lyon C.C. Highet A.S. Successful treatment of pain in two patients with cutaneous leiomyomata with the oral alpha-1 adrenoceptor antagonist, doxazosin.Br J Dermatol. 2004; 150: 770-795Crossref PubMed Scopus (30) Google Scholar The neurotransmitters serotonin (5-HT) and norepinephrine (NE) regulate pain signals in the central nervous system. Dual 5-HT serotonin and NE reuptake inhibitors—including venlafaxine and duloxetine—are being used as antidepressant agents. However, both venlafaxine and duloxetine share an additional therapeutic action as analgesics with more favorable tolerability profiles compared with other therapeutic agents.6Sindrup S.H. Bach F.W. Madsen C. Gram L.F. Jensen T.S. Venlafaxine versus imipramine in painful polyneuropathy: a randomized, controlled trial.Neurology. 2003; 60: 1284-1289Crossref PubMed Scopus (350) Google Scholar, 7Sumpton J.E. Moulin D.E. Treatment of neuropathic pain with venlafaxine.Ann Pharmacother. 2001; 35: 557-559Crossref PubMed Scopus (82) Google Scholar, 8Wernicke J.F. Raskin J. Rosen A. et al.Duloxetine in the long-term management of diabetic peripheral neuropathic pain: an open-label, 52-week extension of a randomized controlled clinical trial.Curr Ther Res. 2006; 67: 283-304Abstract Full Text PDF PubMed Scopus (23) Google Scholar Pregabalin is a structural analog of gamma-aminobutyric acid similar to gabapentin, which has been found effective in neuropathic and nociceptive pain.9Ben-Menachem E. Pregabalin pharmacology and its relevance to clinical practice.Epilepsia. 2004; 45: 13-18Crossref PubMed Scopus (421) Google Scholar In our case, cutaneous leiomyomatosis gradually became symptomatic over years. Treatment with nonsteroidal anti-inflammatory drugs failed and both nifedipine and amitriptyline were stopped because of side effects. Venlafaxine was used in a relatively high daily dose to treat both the patient's pain and depression. Although venlafaxine in combination with pregabalin resulted in pain relief, only a partial improvement of the burning sensation was achieved. However, venlafaxine in high dosage was not tolerated. Pain relief finally was achieved with the combined therapy of pregabalin and duloxetine. To our knowledge, this is the first report of successful pain treatment with duloxetine and pregabalin in a patient with cutaneous leiomyomata." @default.
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• W2093586341 title "Successful Pain Relief of Cutaneous Leiomyomata Due to Reed Syndrome with the Combination Treatment of Pregabalin and Duloxetine" @default.
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