Eur Spine J DOI 10.1007/s00586-013-2988-x
CASE REPORT
Low back pain tied to spinal endometriosis Zhao Dongxu • Yin Fei • Xiao Xing Zhang Bo-Yin • Zhu Qingsan
•
Received: 22 July 2012 / Revised: 28 August 2013 / Accepted: 29 August 2013 Ó Springer-Verlag Berlin Heidelberg 2014
Abstract Study design Case report. Objective We present a case of endometriosis of lumbar vertebrae. The literatures are reviewed with endometriosis of spine. Summary of background data Endometriosis is a common condition, which is defined as endometrial tissue lying outside the endometrial cavity. It is usually found within the peritoneal cavity, predominantly within the pelvis, commonly on the uterosacral ligaments. It can also be found in other sites such as umbilicus, abdominal scars, nasal passages and pleural cavity. But it is very rarely seen in the spine, with no report of endometriosis found in the lumbar vertebrae. Method A 33-year-old woman presented with severe low back pain. She had the low back pain periodically for 3 years, and the pain was associated with menstruation. Radiographs showed a lesion in the posterior L3 body. After surgery, tissue biopsy indicated the presence of endometrial tissue in the lesion and thus confirmed endometriosis. Results Most cases of spine endometriosis that have been reported are usually found inside spinal canal, endorachis or spinal cord. But spinal vertebrae can also be involved in endometriosis. Conclusions Although endometriosis is a rare possible cause of periodical low back pain in women of childbearing age, we suggest that if a woman suffering from periodical low back pain is encountered, do not ignore the possibility of endometriosis in the spine. Z. Dongxu Y. Fei X. Xing Z. Bo-Yin Z. Qingsan (&) Department of Orthopaedic Surgery, China-Japan Union Hospital of Jilin University, Changchun, Jilin 130033, China e-mail: [email protected]
Keywords back pain
Endometriosis Vertebral body Low
Case report A 33-year-old woman complained of having lower back pain periodically for 3 years and the pain was associated with menstruation. The pain aggravated in the last 1 year before admitting to the hospital with additional left lower extremity pain. The low back pain was more severe at the beginning of menstruation than at the end of menstruation. X-Plain film (Fig. 1) showed a hemicycle lesion with calcification at the edge in the posterior of L3 vertebral body. Computed tomography (CT) (Fig. 2) scanning revealed a irregularly destroyed posterior border of L3 vertebrae and ossification was noted in the border of the lesion. The left pedicle had a destroyed lesion. Magnetic resonance imaging (MRI) done during the menstrual period showed markedly increased signal intensity on the T1WI and T2WI in the posterior of L3 vertebral body and the niduses compressed dural sac (Fig. 3). MRI taken in intermenstrual period showed miscellaneous signals which involved high or equal signals on the T2WI that of posterior of L3 vertebral body and left pedicle, while T1WI indicated miscellaneous equal or low signals (Fig. 4). Before surgery, imaging only showed a tumor-like lesion in L3 vertebral body. Tumor resection was carried out to examine the tissue. During the operation, we found that the mass in the L3 posterior border was a ‘‘fish-like’’ tissue with necrosis and hemorrhage and was slightly adhesive with dura sac. We separated the adhesion and removed the mass as thoroughly as we could. The collapsed vertebral body fragments were sent for
123
Eur Spine J
pathologic analysis. Histopathologic examination revealed that the epithelial cells appeared in the glands were in fact columnar. There were signs of hemorrhage and presence of inflammatory cells in the interstitial tissue (Fig. 5). Immunohistochemistry studies indicated that the cells were strongly positive for cytokeratin
(CK7??), estrogen receptor (ER??) and progestin receptor (PR?). After the operation, a permanent therapy of Danazol (400 mg daily) was given for 12 months. There were no recurred symptoms in these 12 months even during menstrual cycles.
Fig. 1 X-Plain film showed a hemicycle lesion with calcification at the edge in the posterior of L3 vertebral body
Fig. 3 Magnetic resonance imaging (MRI) done during the menstrual period showed markedly increased signal intensity on the T1WI and T2WI in the posterior of L3 vertebral body
Fig. 2 Computed tomography (CT) scanning revealed a irregularly destroyed posterior border of L3 vertebrae and ossification was noted in the border of the lesion
123
Eur Spine J
Discussion Endometriosis is defined as the presence of endometrium external to the uterus. The ectopic endometrial tissue is active and responds similarly to the endometrium during the menstrual cycle, resulting in bleeding, inflammation, and scarring [1]. In 2002, US Department of Health and Human Services estimated 5.5 million women in North America, representing 2–10 % of the population, suffered from endometriosis [2]. Women of reproductive age are the most susceptible to endometriosis, with the highest incidence occurring between 25 and 29 years of age [3]. Women with
Fig. 4 MRI taken in intermenstrual period showed miscellaneous signals which involved high or equal signals on the T2WI that of posterior of L3 vertebral body and left pedicle, while T1WI indicated miscellaneous equal or low signals
symptomatic endometriosis typically experience pain in the hypogastric and perineal regions [4], but also can experience pain in the lower back and lower extremities [5, 6]. Low back pain is one of the most common musculoskeletal impairments in physical therapist practice, accounting for 36–53 % of all client visits [7]. Although the prevalence of back pain is low between ages of 25 and 35 years, the incidence of nonspecific low back pain is high in those reproductive women [8, 9]. The incidence and prevalence of endometriosis also are high in the same age range with nonspecific low back pain. So endometriosis presents a challenge to the spinal surgeon during the diagnostic process. In women of reproductive age, it is difficult to distinguish the manifestations of nonspecific low back pain from the low back pain caused by endometriosis in the spine. Imaging such as MRI and CT scanning can be done to exclude a herniated nucleus pulposus and vertebral fracture. MR imaging is now commonly used for diagnosis of endometriosis [10], but MRI scans show a large spectrum of images depending on the day of menstrual cycle. We suggest it is necessary to perform investigational scanning in reproductive women in different periods of menstruation [11]. The initial plan of care is physical therapy. Spinal stabilization exercises also can be introduced in early days [12]. When endometriosis is considered in spine, medical treatment is considered first. If the symptoms such as the low back pain does not alleviate or the side effects of drugs serious or the patient having the desire of pregnancy, the next step is to perform surgery to remove the lesion causing the pain. It also truncates the possibility of compression for spinal cord and nerve trunks. After surgery, medical therapy is also necessary, several different preparations [oral contraceptives, progestogenics, gestrinone, danazol, and gonadotropin releasing hormone (GnRH) agonist] and new options [gonadotropin releasing hormone antagonists (GnRHA), aromatase inhibitors, estrogen receptor beta agonist, progesterone receptor modulators, angiogenesis inhibitors, and COX-2 selective inhibitors] are available [10].
Fig. 5 Pathological slice indicated that there were tissue hemorrhage and of inflammatory cells in the interstitial tissue
HE,
4
HE,
20
123
Eur Spine J
If pain symptoms continue to persist after attempting medical and surgical management, or side effects from medical therapy become intolerable, definitive surgery, which includes hysterectomy with bilateral salpingectomy and oophorectomy, may be considered as a final treatment option in patients with no plans for pregnancy [13]. Patients should understand that even after definitive surgery, endometriosis may recur in 5–15 % of cases [14], and even can lead to malignant transformation [15].
Conclusions Endometriosis, in which common symptoms are nonspecific and frequently present with normal physical examination findings, can be difficult to diagnose. If misdiagnosed and left untreated, endometriosis can progress from a small lesion to a larger one with extensive fibrosis and adhesions. In spine this can further lead to significant distortion of the spinal anatomy and structure, thus developing into a more severe deeply infiltrating stage of the disease that may affect spinal cord and roots. In our opinion, when conventional therapies fail to resolve the cyclic low back pain in fertile women, spinal endometriosis must be recognized as a potential cause of periodic neurological signs and symptoms especially in young and middle-aged women. MRI of the lumbosacral spine is necessary. The choices of treatment depend on the pain or symptoms and the patient’s future plans for pregnancy. Attempts at total removal of endometriosis in the spine may be necessary. Conflict of interest interest.
None of the authors has any potential conflict of
References 1. Murphy AA (2002) Clinical aspects of endometriosis. Ann N Y Acad Sci 955:1–10
123
2. National Institutes of Health (2002) Endometriosis, NIH Publication 02-2413 National Institute of Child Health and Human Development, US Department of Health and Human Services, Rockville, MD 3. Missmer SA, Hankinson SE, Spiegelman D et al (2004) Incidence of laparoscopically confirmed endometriosis by demographic, anthropometric, and lifestyle factors. Am J Epidemiol 160:784–796 4. Koninckx PR, Meuleman C, Oosterlynck D, Cornillie FJ (1996) Diagnosis of deep endometriosis by clinical examination during menstruation and plasma CA-125 concentration. Fertil Steril 65:280–287 5. Macek C (1983) Neurological deficits, back pain tied to endometriosis. JAMA 249:686–688 6. Prendergast SA, Weiss JM (2003) Screening for musculoskeletal causes of pelvic pain. Clin Obstet Gynecol 46:773–782 7. Battie´ MC, Cherkin DC, Dunn R et al (1994) Managing low back pain: attitudes and treatment preferences of physical therapists. Phys Ther 74:219–226 8. Taguchi T (2003) Low back pain in young and middle-aged people. Jpn Med Assoc J 46:417–423 9. Loney PL, Stratford PW (1999) The prevalence of low back pain in adults: a methodological review of the literature. Phys Ther 79:384–396 10. Togashi K, Nishimura K, Kimura I, Tsuda Y, Yamashita K, Shibata T et al (1991) Endometrial cysts: diagnosis with MR imaging. Radiology 180:73–78 11. Balleyguier C, Chapron C, Dubuisson JB, Kinkel K, Fauconnier A, Vieira M et al (2002) Comparison of magnetic resonance imaging and transvaginal ultrasonography in diagnosing bladder endometriosis. J Am Assoc Gynecol Laparosc 9:15–23 12. Delitto A, Erhand RE, Bowling RW (1995) A treatment-based classification approach to low back syndrome: identifying and staging patients for conservative treatment. Phys Ther 75:470–485 13. Mounsey AL, Wilgus A, Slawson DC (2006) Diagnosis and management of endometriosis. Am Fam Physician 74(4):594–600 14. Jackson B, Telner DE (2006) Managing the misplaced: approach to endometriosis. Can Fam Physician 52(11):1420–1424 15. Richter K (1977) Endometrioid carcinoma of the spinal canal. Geburtshilfe Frauenheilkd 37(9):771–775
CASE REPORT
Low back pain tied to spinal endometriosis Zhao Dongxu • Yin Fei • Xiao Xing Zhang Bo-Yin • Zhu Qingsan
•
Received: 22 July 2012 / Revised: 28 August 2013 / Accepted: 29 August 2013 Ó Springer-Verlag Berlin Heidelberg 2014
Abstract Study design Case report. Objective We present a case of endometriosis of lumbar vertebrae. The literatures are reviewed with endometriosis of spine. Summary of background data Endometriosis is a common condition, which is defined as endometrial tissue lying outside the endometrial cavity. It is usually found within the peritoneal cavity, predominantly within the pelvis, commonly on the uterosacral ligaments. It can also be found in other sites such as umbilicus, abdominal scars, nasal passages and pleural cavity. But it is very rarely seen in the spine, with no report of endometriosis found in the lumbar vertebrae. Method A 33-year-old woman presented with severe low back pain. She had the low back pain periodically for 3 years, and the pain was associated with menstruation. Radiographs showed a lesion in the posterior L3 body. After surgery, tissue biopsy indicated the presence of endometrial tissue in the lesion and thus confirmed endometriosis. Results Most cases of spine endometriosis that have been reported are usually found inside spinal canal, endorachis or spinal cord. But spinal vertebrae can also be involved in endometriosis. Conclusions Although endometriosis is a rare possible cause of periodical low back pain in women of childbearing age, we suggest that if a woman suffering from periodical low back pain is encountered, do not ignore the possibility of endometriosis in the spine. Z. Dongxu Y. Fei X. Xing Z. Bo-Yin Z. Qingsan (&) Department of Orthopaedic Surgery, China-Japan Union Hospital of Jilin University, Changchun, Jilin 130033, China e-mail: [email protected]
Keywords back pain
Endometriosis Vertebral body Low
Case report A 33-year-old woman complained of having lower back pain periodically for 3 years and the pain was associated with menstruation. The pain aggravated in the last 1 year before admitting to the hospital with additional left lower extremity pain. The low back pain was more severe at the beginning of menstruation than at the end of menstruation. X-Plain film (Fig. 1) showed a hemicycle lesion with calcification at the edge in the posterior of L3 vertebral body. Computed tomography (CT) (Fig. 2) scanning revealed a irregularly destroyed posterior border of L3 vertebrae and ossification was noted in the border of the lesion. The left pedicle had a destroyed lesion. Magnetic resonance imaging (MRI) done during the menstrual period showed markedly increased signal intensity on the T1WI and T2WI in the posterior of L3 vertebral body and the niduses compressed dural sac (Fig. 3). MRI taken in intermenstrual period showed miscellaneous signals which involved high or equal signals on the T2WI that of posterior of L3 vertebral body and left pedicle, while T1WI indicated miscellaneous equal or low signals (Fig. 4). Before surgery, imaging only showed a tumor-like lesion in L3 vertebral body. Tumor resection was carried out to examine the tissue. During the operation, we found that the mass in the L3 posterior border was a ‘‘fish-like’’ tissue with necrosis and hemorrhage and was slightly adhesive with dura sac. We separated the adhesion and removed the mass as thoroughly as we could. The collapsed vertebral body fragments were sent for
123
Eur Spine J
pathologic analysis. Histopathologic examination revealed that the epithelial cells appeared in the glands were in fact columnar. There were signs of hemorrhage and presence of inflammatory cells in the interstitial tissue (Fig. 5). Immunohistochemistry studies indicated that the cells were strongly positive for cytokeratin
(CK7??), estrogen receptor (ER??) and progestin receptor (PR?). After the operation, a permanent therapy of Danazol (400 mg daily) was given for 12 months. There were no recurred symptoms in these 12 months even during menstrual cycles.
Fig. 1 X-Plain film showed a hemicycle lesion with calcification at the edge in the posterior of L3 vertebral body
Fig. 3 Magnetic resonance imaging (MRI) done during the menstrual period showed markedly increased signal intensity on the T1WI and T2WI in the posterior of L3 vertebral body
Fig. 2 Computed tomography (CT) scanning revealed a irregularly destroyed posterior border of L3 vertebrae and ossification was noted in the border of the lesion
123
Eur Spine J
Discussion Endometriosis is defined as the presence of endometrium external to the uterus. The ectopic endometrial tissue is active and responds similarly to the endometrium during the menstrual cycle, resulting in bleeding, inflammation, and scarring [1]. In 2002, US Department of Health and Human Services estimated 5.5 million women in North America, representing 2–10 % of the population, suffered from endometriosis [2]. Women of reproductive age are the most susceptible to endometriosis, with the highest incidence occurring between 25 and 29 years of age [3]. Women with
Fig. 4 MRI taken in intermenstrual period showed miscellaneous signals which involved high or equal signals on the T2WI that of posterior of L3 vertebral body and left pedicle, while T1WI indicated miscellaneous equal or low signals
symptomatic endometriosis typically experience pain in the hypogastric and perineal regions [4], but also can experience pain in the lower back and lower extremities [5, 6]. Low back pain is one of the most common musculoskeletal impairments in physical therapist practice, accounting for 36–53 % of all client visits [7]. Although the prevalence of back pain is low between ages of 25 and 35 years, the incidence of nonspecific low back pain is high in those reproductive women [8, 9]. The incidence and prevalence of endometriosis also are high in the same age range with nonspecific low back pain. So endometriosis presents a challenge to the spinal surgeon during the diagnostic process. In women of reproductive age, it is difficult to distinguish the manifestations of nonspecific low back pain from the low back pain caused by endometriosis in the spine. Imaging such as MRI and CT scanning can be done to exclude a herniated nucleus pulposus and vertebral fracture. MR imaging is now commonly used for diagnosis of endometriosis [10], but MRI scans show a large spectrum of images depending on the day of menstrual cycle. We suggest it is necessary to perform investigational scanning in reproductive women in different periods of menstruation [11]. The initial plan of care is physical therapy. Spinal stabilization exercises also can be introduced in early days [12]. When endometriosis is considered in spine, medical treatment is considered first. If the symptoms such as the low back pain does not alleviate or the side effects of drugs serious or the patient having the desire of pregnancy, the next step is to perform surgery to remove the lesion causing the pain. It also truncates the possibility of compression for spinal cord and nerve trunks. After surgery, medical therapy is also necessary, several different preparations [oral contraceptives, progestogenics, gestrinone, danazol, and gonadotropin releasing hormone (GnRH) agonist] and new options [gonadotropin releasing hormone antagonists (GnRHA), aromatase inhibitors, estrogen receptor beta agonist, progesterone receptor modulators, angiogenesis inhibitors, and COX-2 selective inhibitors] are available [10].
Fig. 5 Pathological slice indicated that there were tissue hemorrhage and of inflammatory cells in the interstitial tissue
HE,
4
HE,
20
123
Eur Spine J
If pain symptoms continue to persist after attempting medical and surgical management, or side effects from medical therapy become intolerable, definitive surgery, which includes hysterectomy with bilateral salpingectomy and oophorectomy, may be considered as a final treatment option in patients with no plans for pregnancy [13]. Patients should understand that even after definitive surgery, endometriosis may recur in 5–15 % of cases [14], and even can lead to malignant transformation [15].
Conclusions Endometriosis, in which common symptoms are nonspecific and frequently present with normal physical examination findings, can be difficult to diagnose. If misdiagnosed and left untreated, endometriosis can progress from a small lesion to a larger one with extensive fibrosis and adhesions. In spine this can further lead to significant distortion of the spinal anatomy and structure, thus developing into a more severe deeply infiltrating stage of the disease that may affect spinal cord and roots. In our opinion, when conventional therapies fail to resolve the cyclic low back pain in fertile women, spinal endometriosis must be recognized as a potential cause of periodic neurological signs and symptoms especially in young and middle-aged women. MRI of the lumbosacral spine is necessary. The choices of treatment depend on the pain or symptoms and the patient’s future plans for pregnancy. Attempts at total removal of endometriosis in the spine may be necessary. Conflict of interest interest.
None of the authors has any potential conflict of
References 1. Murphy AA (2002) Clinical aspects of endometriosis. Ann N Y Acad Sci 955:1–10
123
2. National Institutes of Health (2002) Endometriosis, NIH Publication 02-2413 National Institute of Child Health and Human Development, US Department of Health and Human Services, Rockville, MD 3. Missmer SA, Hankinson SE, Spiegelman D et al (2004) Incidence of laparoscopically confirmed endometriosis by demographic, anthropometric, and lifestyle factors. Am J Epidemiol 160:784–796 4. Koninckx PR, Meuleman C, Oosterlynck D, Cornillie FJ (1996) Diagnosis of deep endometriosis by clinical examination during menstruation and plasma CA-125 concentration. Fertil Steril 65:280–287 5. Macek C (1983) Neurological deficits, back pain tied to endometriosis. JAMA 249:686–688 6. Prendergast SA, Weiss JM (2003) Screening for musculoskeletal causes of pelvic pain. Clin Obstet Gynecol 46:773–782 7. Battie´ MC, Cherkin DC, Dunn R et al (1994) Managing low back pain: attitudes and treatment preferences of physical therapists. Phys Ther 74:219–226 8. Taguchi T (2003) Low back pain in young and middle-aged people. Jpn Med Assoc J 46:417–423 9. Loney PL, Stratford PW (1999) The prevalence of low back pain in adults: a methodological review of the literature. Phys Ther 79:384–396 10. Togashi K, Nishimura K, Kimura I, Tsuda Y, Yamashita K, Shibata T et al (1991) Endometrial cysts: diagnosis with MR imaging. Radiology 180:73–78 11. Balleyguier C, Chapron C, Dubuisson JB, Kinkel K, Fauconnier A, Vieira M et al (2002) Comparison of magnetic resonance imaging and transvaginal ultrasonography in diagnosing bladder endometriosis. J Am Assoc Gynecol Laparosc 9:15–23 12. Delitto A, Erhand RE, Bowling RW (1995) A treatment-based classification approach to low back syndrome: identifying and staging patients for conservative treatment. Phys Ther 75:470–485 13. Mounsey AL, Wilgus A, Slawson DC (2006) Diagnosis and management of endometriosis. Am Fam Physician 74(4):594–600 14. Jackson B, Telner DE (2006) Managing the misplaced: approach to endometriosis. Can Fam Physician 52(11):1420–1424 15. Richter K (1977) Endometrioid carcinoma of the spinal canal. Geburtshilfe Frauenheilkd 37(9):771–775
