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Endometriosis of the soleus and gastrocnemius muscles
CASE REPORT Endometriosis of the soleus and gastrocnemius muscles Omero Benedicto Poli-Neto, M.D.,a,b J ulio C esar Rosa-e-Silva, M.D.,b Hermes Freitas Barbosa, M.D.,b b Francisco Jos e Candido-dos-Reis, M.D., and Antonio Alberto Nogueira, M.D.b a
Department of Surgery and Anatomy and b Department of Gynecology and Obstetrics, Faculty of Medicine, University of S~ao Paulo, Ribeir~ao Preto, Brazil
Objective: To document a rare case of endometriosis in the soleus and gastrocnemius muscles. Design: Case report. Setting: Tertiary care center. Patient(s): A 30-year-old fertile woman presented with moderate dysmenorrhea associated with calf pain and bulging that had been gradually worsening over the last years, particularly during menses. A mass in the soleus and gastrocnemius muscles was identified in ultrasonography and magnetic resonance imaging. Intervention(s): Endometriosis was diagnosed by incisional biopsy on the basis of histopathology, and wide excisional biopsy was performed. Main Outcome Measure(s): Unusual clinical presentation of endometriosis. Result(s): The patient was disease free for 2 months. Recurrence of the lesion was then diagnosed, and a new surgical approach was planned. Conclusion(s): Endometriosis in muscles is a rare event, and existing theories are not totally sufficient in explaining it. (Fertil Steril 2009;91:1294.e13–e15. 2009 by American Society for Reproductive Medicine.) Key Words: Endometriosis, musculoskeletal system, soleus muscle, gastrocnemius muscle, dysmenorrhea, pain
Endometriosis is characterized by the aberrant presence of endometrial tissue outside the uterine cavity. The prevalence of pelvic endometriosis is 6%–10% among women of reproductive age (1), but extrapelvic disease is less common. Muscular endometriosis is a rare condition, and few cases without surgical implantation have been identified in the literature (2–6). The present case report documents a rare case of endometriosis of the soleus and gastrocnemius muscles associated with moderate dysmenorrhea. To our knowledge, it is the first such case reported.
CASE REPORT The patient was a 30-year-old woman, body mass index (BMI) 23.4 kg/m2 (weight 55.4 kg, height 1.54 m), G3P3 (a vaginal delivery plus episiotomy and two cesarean sections at the age of 18, 20, and 28 years, respectively). menarche at the age of 14 years, regular menses (duration 3 days, interval
Received September 1, 2008; revised November 3, 2008; accepted December 10, 2008; published online January 18, 2009. O.B.P.-N. has nothing to disclose. J.C.R.-e-S. has nothing to disclose. H.d.-F.B. has nothing to disclose. F.J.C.-d.-R. has nothing to disclose. A.A.N. has nothing to disclose. Reprint requests: Omero Benedicto Poli Neto, Av Bandeirantes, ~o Preto—Sa ~o Paulo, Brazil 3900—8 andar, CEP 14049-900, Ribeira (FAX: þ551636330946; E-mail: [email protected]).
28 days on average). She had been using ethinylestradiol 0.02 mg plus gestodene 0.075 mg for 2 years. Her clinical history included moderate dysmenorrhea (affecting daily activity and responsive to medication, rarely causing absenteeism) but not dispareunia, associated with pain and bulging of the calf that had been gradually worsening over the last 8 years. The patient felt more pronounced pain at the time of the menses. Physical examination revealed a hardened, painful, and ill defined tumor growth in the median region of the right calf measuring 10 cm at its widest diameter. Muscle strength and associated musculature were preserved. Gynecologic examination revealed no pelvic abnormalities. Ultrasonography demonstrated a heterogeneous predominantly hypoechoic tumor measuring 2.6 0.9 1.3 cm in the soleus and gastrocnemius muscles (Fig. 1A). On magnetic resonance imaging (MRI), the mass showed a serpiginous aspect (4.5 2.1 cm), with a low T1 signal (Fig. 1B) and a high T2 signal (Fig. 1C). There was no significant difference between pre- and postcontrast MRI (Figs. 1D and 1E). Transvaginal pelvic ultrasonography did not show alterations. The CA-125 level was 6.74 U/mL. An incision biopsy was performed in the right calf, and histopathologic examination revealed an endometriotic nodule (Fig. 1F). Wide surgical excision was performed, but the lesion returned and at the time of writing, a new surgical
1294.e13 Fertility and Sterility Vol. 91, No. 4, April 2009 Copyright ª2009 American Society for Reproductive Medicine, Published by Elsevier Inc.
0015-0282/09/$36.00 doi:10.1016/j.fertnstert.2008.12.014
FIGURE 1 (A) Ultrasound image revealing a heterogeneous predominantly hypoechoic mass of imprecise limits (arrows) measuring 2.6 0.9 1.3 cm; (B) Fast spin-echo T1-weighted axial magnetic resonance image (MRI) shows that the mass has a low signal intensity and is almost as isointense as muscle; (C) Fast spin-echo T2-weighted axial MRI with fat saturation shows that the gastrocnemius (medial head) and soleus mass (arrows) have a high signal intensity; (D) Fast spin-echo T1-weighted coronal MRI precontrast mass (arrows); (E) Fast spin-echo T1weighted coronal MR image postcontrast mass (arrows); (F) Endometriotic nodule composed of clusters of endometrial epithelial cells in a dense fibroadipose tissue, lymphocytes, and plasmocytes (hematoxylin-eosin).
Poli-Neto. A rare case of endometriosis at the leg. Fertil Steril 2009.
approach is being considered for treatment. Initially, we did not indicate diagnostic laparoscopy, because of little evidence of peritoneal disease and because of the patient’s wish to receive only local treatment.
DISCUSSION Extrapelvic endometriosis is much less common than intrapelvic endometriosis and often involves considerable diagnostic confusion. The explanation for the occurrence of endometriosis at this site is not supported by more generally accepted theories, i.e., retrograde menstruation (7), coelomic metaplasia (8), or Fertility and Sterility
immunologic explanations (9). In addition, iatrogenic deposition of endometrial tissue in surgical scar (10) does not explain the present case. The potential of autochthonous endometrial cells for lymphatic and/or vascular dissemination (11) is the most plausible factor explaining the occurrence of the type of lesion detected here, as also postulated by other authors in similar situations (4). We cannot draw any conclusions regarding the role of cesarean section as an element facilitating the onset of the lesion, although the patient was submitted to a cesarean section before the onset of symptoms. The influence of environmental factors may also play a relevant role (12). However, in view of the singularity of the situation, it is difficult to establish any associations. 1294.e14
In conclusion, endometriosis is a complex disease with poorly understood physiopathology and with multiple clinical expressions. Existing theories are not sufficient in explaining it and the professional’s perspicacity is essential for correct diagnosis. REFERENCES 1. Strathy JH, Molgaard CA, Coulam CB, Melton LJ 3rd. Endometriosis and infertility: a laparoscopic study of endometriosis among fertile and infertile women. Fertil Steril 1982;38:667–72. 2. Gennari L, Luciani L. [A case of endometriosis of the trapezius muscle]. Tumori 1965;51:361–5. Italian. 3. Aron SE. [Endometriosis in the region of the deltoid muscle.]. Arkh Patol 1957;19:67–8. Russian. 4. Basu PA, Kesani AK, Stacy GS, Peabody TD. Endometriosis of the vastus lateralis muscle. Skeletal Radiol 2006;35:595–8. 5. Crespo R, Puig F, Marquina I. Pyramidalis muscle endometriosis in absence of previous surgery. Int J Gynaecol Obstet 2005;89:148–9.
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Poli-Neto et al.
6. Hickey NA, Murphy JP, Bloom C, Hamilton P. Magnetic resonance imaging of endometriosis of the piriform muscle causing sciatica: case report. Can Assoc Radiol J 1999;50:33–6. 7. Sampson JA. Peritoneal endometriosis due to menstrual dissemination of endometrial tissue into the peritoneal cavity. Am J Obstet Gynecol 1927;14:422–69. 8. Suginami H. A reappraisal of the coelomic metaplasia theory by reviewing endometriosis occurring in unusual sites and instances. Am J Obstet Gynecol 1991;165:214–8. 9. Matarese G, De Placido G, Nikas Y, Alviggi C. Pathogenesis of endometriosis: natural immunity dysfunction or autoimmune disease? Trends Mol Med 2003;9:223–8. 10. Brenner C, Wohlgemuth S. Scar endometriosis. Surg Gynecol Obstet 1990;170:538–40. 11. Halban J. Histeroadenosis metastatica. Die linphogene genese der sog. Adenofibromatosis heterotopica [German]. Arch Gynak 1925;124: 457–82. 12. Harrad S, Wang Y, Sandaradura S, Leeds A. Human dietary intake and excretion of dioxin-like compounds. J Environ Monit 2003;5: 224–8.
A rare case of endometriosis at the leg
Vol. 91, No. 4, April 2009
Department of Surgery and Anatomy and b Department of Gynecology and Obstetrics, Faculty of Medicine, University of S~ao Paulo, Ribeir~ao Preto, Brazil
Objective: To document a rare case of endometriosis in the soleus and gastrocnemius muscles. Design: Case report. Setting: Tertiary care center. Patient(s): A 30-year-old fertile woman presented with moderate dysmenorrhea associated with calf pain and bulging that had been gradually worsening over the last years, particularly during menses. A mass in the soleus and gastrocnemius muscles was identified in ultrasonography and magnetic resonance imaging. Intervention(s): Endometriosis was diagnosed by incisional biopsy on the basis of histopathology, and wide excisional biopsy was performed. Main Outcome Measure(s): Unusual clinical presentation of endometriosis. Result(s): The patient was disease free for 2 months. Recurrence of the lesion was then diagnosed, and a new surgical approach was planned. Conclusion(s): Endometriosis in muscles is a rare event, and existing theories are not totally sufficient in explaining it. (Fertil Steril 2009;91:1294.e13–e15. 2009 by American Society for Reproductive Medicine.) Key Words: Endometriosis, musculoskeletal system, soleus muscle, gastrocnemius muscle, dysmenorrhea, pain
Endometriosis is characterized by the aberrant presence of endometrial tissue outside the uterine cavity. The prevalence of pelvic endometriosis is 6%–10% among women of reproductive age (1), but extrapelvic disease is less common. Muscular endometriosis is a rare condition, and few cases without surgical implantation have been identified in the literature (2–6). The present case report documents a rare case of endometriosis of the soleus and gastrocnemius muscles associated with moderate dysmenorrhea. To our knowledge, it is the first such case reported.
CASE REPORT The patient was a 30-year-old woman, body mass index (BMI) 23.4 kg/m2 (weight 55.4 kg, height 1.54 m), G3P3 (a vaginal delivery plus episiotomy and two cesarean sections at the age of 18, 20, and 28 years, respectively). menarche at the age of 14 years, regular menses (duration 3 days, interval
Received September 1, 2008; revised November 3, 2008; accepted December 10, 2008; published online January 18, 2009. O.B.P.-N. has nothing to disclose. J.C.R.-e-S. has nothing to disclose. H.d.-F.B. has nothing to disclose. F.J.C.-d.-R. has nothing to disclose. A.A.N. has nothing to disclose. Reprint requests: Omero Benedicto Poli Neto, Av Bandeirantes, ~o Preto—Sa ~o Paulo, Brazil 3900—8 andar, CEP 14049-900, Ribeira (FAX: þ551636330946; E-mail: [email protected]).
28 days on average). She had been using ethinylestradiol 0.02 mg plus gestodene 0.075 mg for 2 years. Her clinical history included moderate dysmenorrhea (affecting daily activity and responsive to medication, rarely causing absenteeism) but not dispareunia, associated with pain and bulging of the calf that had been gradually worsening over the last 8 years. The patient felt more pronounced pain at the time of the menses. Physical examination revealed a hardened, painful, and ill defined tumor growth in the median region of the right calf measuring 10 cm at its widest diameter. Muscle strength and associated musculature were preserved. Gynecologic examination revealed no pelvic abnormalities. Ultrasonography demonstrated a heterogeneous predominantly hypoechoic tumor measuring 2.6 0.9 1.3 cm in the soleus and gastrocnemius muscles (Fig. 1A). On magnetic resonance imaging (MRI), the mass showed a serpiginous aspect (4.5 2.1 cm), with a low T1 signal (Fig. 1B) and a high T2 signal (Fig. 1C). There was no significant difference between pre- and postcontrast MRI (Figs. 1D and 1E). Transvaginal pelvic ultrasonography did not show alterations. The CA-125 level was 6.74 U/mL. An incision biopsy was performed in the right calf, and histopathologic examination revealed an endometriotic nodule (Fig. 1F). Wide surgical excision was performed, but the lesion returned and at the time of writing, a new surgical
1294.e13 Fertility and Sterility Vol. 91, No. 4, April 2009 Copyright ª2009 American Society for Reproductive Medicine, Published by Elsevier Inc.
0015-0282/09/$36.00 doi:10.1016/j.fertnstert.2008.12.014
FIGURE 1 (A) Ultrasound image revealing a heterogeneous predominantly hypoechoic mass of imprecise limits (arrows) measuring 2.6 0.9 1.3 cm; (B) Fast spin-echo T1-weighted axial magnetic resonance image (MRI) shows that the mass has a low signal intensity and is almost as isointense as muscle; (C) Fast spin-echo T2-weighted axial MRI with fat saturation shows that the gastrocnemius (medial head) and soleus mass (arrows) have a high signal intensity; (D) Fast spin-echo T1-weighted coronal MRI precontrast mass (arrows); (E) Fast spin-echo T1weighted coronal MR image postcontrast mass (arrows); (F) Endometriotic nodule composed of clusters of endometrial epithelial cells in a dense fibroadipose tissue, lymphocytes, and plasmocytes (hematoxylin-eosin).
Poli-Neto. A rare case of endometriosis at the leg. Fertil Steril 2009.
approach is being considered for treatment. Initially, we did not indicate diagnostic laparoscopy, because of little evidence of peritoneal disease and because of the patient’s wish to receive only local treatment.
DISCUSSION Extrapelvic endometriosis is much less common than intrapelvic endometriosis and often involves considerable diagnostic confusion. The explanation for the occurrence of endometriosis at this site is not supported by more generally accepted theories, i.e., retrograde menstruation (7), coelomic metaplasia (8), or Fertility and Sterility
immunologic explanations (9). In addition, iatrogenic deposition of endometrial tissue in surgical scar (10) does not explain the present case. The potential of autochthonous endometrial cells for lymphatic and/or vascular dissemination (11) is the most plausible factor explaining the occurrence of the type of lesion detected here, as also postulated by other authors in similar situations (4). We cannot draw any conclusions regarding the role of cesarean section as an element facilitating the onset of the lesion, although the patient was submitted to a cesarean section before the onset of symptoms. The influence of environmental factors may also play a relevant role (12). However, in view of the singularity of the situation, it is difficult to establish any associations. 1294.e14
In conclusion, endometriosis is a complex disease with poorly understood physiopathology and with multiple clinical expressions. Existing theories are not sufficient in explaining it and the professional’s perspicacity is essential for correct diagnosis. REFERENCES 1. Strathy JH, Molgaard CA, Coulam CB, Melton LJ 3rd. Endometriosis and infertility: a laparoscopic study of endometriosis among fertile and infertile women. Fertil Steril 1982;38:667–72. 2. Gennari L, Luciani L. [A case of endometriosis of the trapezius muscle]. Tumori 1965;51:361–5. Italian. 3. Aron SE. [Endometriosis in the region of the deltoid muscle.]. Arkh Patol 1957;19:67–8. Russian. 4. Basu PA, Kesani AK, Stacy GS, Peabody TD. Endometriosis of the vastus lateralis muscle. Skeletal Radiol 2006;35:595–8. 5. Crespo R, Puig F, Marquina I. Pyramidalis muscle endometriosis in absence of previous surgery. Int J Gynaecol Obstet 2005;89:148–9.
1294.e15
Poli-Neto et al.
6. Hickey NA, Murphy JP, Bloom C, Hamilton P. Magnetic resonance imaging of endometriosis of the piriform muscle causing sciatica: case report. Can Assoc Radiol J 1999;50:33–6. 7. Sampson JA. Peritoneal endometriosis due to menstrual dissemination of endometrial tissue into the peritoneal cavity. Am J Obstet Gynecol 1927;14:422–69. 8. Suginami H. A reappraisal of the coelomic metaplasia theory by reviewing endometriosis occurring in unusual sites and instances. Am J Obstet Gynecol 1991;165:214–8. 9. Matarese G, De Placido G, Nikas Y, Alviggi C. Pathogenesis of endometriosis: natural immunity dysfunction or autoimmune disease? Trends Mol Med 2003;9:223–8. 10. Brenner C, Wohlgemuth S. Scar endometriosis. Surg Gynecol Obstet 1990;170:538–40. 11. Halban J. Histeroadenosis metastatica. Die linphogene genese der sog. Adenofibromatosis heterotopica [German]. Arch Gynak 1925;124: 457–82. 12. Harrad S, Wang Y, Sandaradura S, Leeds A. Human dietary intake and excretion of dioxin-like compounds. J Environ Monit 2003;5: 224–8.
A rare case of endometriosis at the leg
Vol. 91, No. 4, April 2009