Current Orthopaedic Practice  

      Issue: Volume 27(3), May/June 2016, p E16-E19
      Copyright: Copyright (C) 2016 Wolters Kluwer Health, Inc. All rights
reserved
      Publication Type: [CASE REPORT]
      DOI: 10.1097/BCO.0000000000000362
      ISSN: 1940-7041
      Accession: 01337441-201605000-00021       

 [CASE REPORT]

  Ischiopubic rami excision for obstructive dyspareunia in hyperparathyroidism  

  Khedr, Ahmed MD, (M.Sc Orthopedics); Khaled, Sherif A. MD, (PhD Orthopedics)  

    Author Information 

  Cairo University Hospitals, Cairo, Egypt  

  Financial Disclosure: The authors have no disclosures and report no conflicts
of interest.  

  Correspondence to Ahmed Khedr, MD, (M.Sc Orthopedics), Orthopedics, Cairo
University, 4 Hosni Metwali street - Elharam, Giza, Egypt 12111 Tel: +20201093944334;
fax: +20235856593; e-mail: ahmedkhedr@kasralainy.edu.eg.  

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    Outline

    INTRODUCTION

    CASE REPORT

    DISCUSSION

    REFERENCES

INTRODUCTION

Hyperparathyroidism was first described in 1891 by Von Recklinghausen.1 Since
that time the mnemonic "stones, bones, abdominal groans, thrones, and psychiatric
overtones" were used to describe its manifestations.2 Hyperparathyroidism
classically produced osteitis fibrosa cystic and pathological fractures.
However, this is not the common presentation of the disease nowadays.1
Nevertheless, there is an increasing interest in the disease, with a larger
number of cases being diagnosed with subtle manifestations as neuropsychiatric
and cardiac symptoms. Furthermore, a newer entity of the disease is being
identified, although its natural history is not fully understood, which is
normocalcemic primary hyperparathyroidism.2

Parathyroid hormone mediates its effect through cAMP as a second messenger. The
hormone acts on PTH-1R receptor, which is present mainly on osteoblasts and in
fewer amounts on osteoclasts.3 Osteoblasts also activate osteoclastic proliferation
and bone resorption.4 The disease causes loss of bone density in cortical sites,
such as the forearm, more than at cancellous sites, such as the hip or spine.1
Therefore, parathyroid hormone is believed to have various anabolic and
catabolic effects on bone, depending on the structure of bone (i.e. cortical or
cancellous) and whether the stimulation of bone by parathyroid hormone is
continuous or intermittent.5-7 This results in the different bony changes and
deformities as seen in our patient.

In this case report, we describe a patient with an unusual presentation of
hyperparathyroidism, namely obstructive dyspareunia from pelvic deformity,
enlargement, and malrotation of the inferior pubic and ischial rami. Informed
consent was obtained by the patient for the procedure and for this report. The
report was approved by the ethical committee of our institution.

CASE REPORT

A 35-year-old woman, married, with a 7-year-old child, presented to the clinic
with obstructive dyspareunia of 1-year duration. The patient was diagnosed as
having primary hyperparathyroidism 8 mo before presenting to our clinic. At
presentation, she was complaining of bony pain and failure of normal marital
sexual intercourse for 1 yr. During this period, multiple investigations were
done, the patient's parathyroid hormone level was 1436 pg/mL. 99mTc-Sestamibi
scan showed no adenoma in the parathyroid gland. The patient did not have other
manifestations of hyperparathyroidism such as pathological fractures, nephrolithiasis,
nephrocalcinosis, constipation, peptic ulcer, pancreatitis, or other cardiovascular
or neuropsychiatric disorders. However, the patient complained of dyspareunia
and difficulty in passage of menstrual flow. The patient reported that she had
to adopt a special position to allow her menses to flow. Plain radiographs
showed trefoil deformity of the pelvis with enlargement of the inferior pubic
and ischial rami (Figure 1). CT showed obstruction of the pelvic outlet by the
enlarged and deformed rami (Figure 2). Gynecological consultation was sought at
this point. The patient reported a history of normal vaginal delivery 7 yr ago
of her only daughter. On vaginal examination, the vagina would not admit the tip
of the little finger. However, pelvic ultrasound did not show any abnormalities
in the reproductive organs.

After planning osteotomy of the ischiopubic rami, the patient was scheduled for
surgery. The patient was placed in the lithotomy position after general
anesthesia. Intravenous prophylactic antibiotics were given. The vagina was
prepped and draped and a urinary catheter was inserted to facilitate identification
and protection of the urethra during surgery. The surgical approach used was
similar to the approach described by Radley, Liebig, and Brown 8 for resection
of the pubis and ischium for treating bone tumors, chronic osteomyelitis, or
tuberculosis. However, as the plan here was not to excise the whole ischium and
pubis, we used only part of the approach. The ischial tuberosities, the inferior
pubic rami, and inferior border of the pubis were marked. The incision was made
in the skin and subcutaneous tissue just lateral to the labia. The obturator
externus, gracilis, adductor brevis and magnus were detached from the outer
surface of the bone, and careful subperiosteal dissection of the ischiocavernosus,
superficial and deep transverse perineal muscles, and obturator internus was
carried out. Retractors were placed to protect the soft tissue, and the
ischiopubic ramus was resected by a sharp small osteotomy. Similarly, the
contralateral ischiopubic ramus was excised. The adequacy of the resection was
checked by vaginal examination and the possibility to introduce 3 fingers
simultaneously. Proper hemostasis was done followed by closure in layers by
absorbable sutures (Figure 3).

The patient's dressing was soaked on the second day after surgery and dressing
was done. Postoperative radiographs were obtained (Figure 4), and the patient
was instructed to remove the dressing on the third postoperative day and perform
perineal washing and cleaning daily. She was instructed to start pelvic floor
strengthening physiotherapy. The patient was evaluated 15 days after surgery.
The wounds had healed with no gapping or infection. Vaginal examination could
admit 3 fingers. The patient had free flow of menstrual bleeding after surgery
and 1 mo later the patient could have sexual intercourse with no pain.

DISCUSSION

Several studies described the effect of the change in shape of the pelvis on
female sexual function and vaginal delivery. These changes are frequently caused
by fractures of the pelvis. The effects of parathyroid hormone on bone
metabolism have been discussed in the literature over the last hundred years.
Mandl in Austria was the first to prove that the enlarged parathyroid was
responsible for the skeletal manifestations of the disease after the first
successful removal of parathyroid adenoma.1 Recently, parathyroid hormone is
believed to have both catabolic and anabolic effects on bone.6

Vallier et al.9 conducted a prospective cohort study in a level 1 trauma center.
One hundred eighty-seven women younger than 55 yr with pelvic fractures were
included. The study concluded that women with pelvic ring injuries report
dyspareunia more than females with other musculoskeletal injuries. Moreover,
symphyseal disruption, symphyseal plating, and alignment greater than 5 mm from
anatomic were associated with dyspareunia. The authors related dyspareunia to
bony changes as well as damage to soft tissues, vascular, and neurological
injuries. The reported incidence of dyspareunia was 78% in patients with type B
injury and 43% in patients with type C injury. Harvey-Kelly et al.10 reviewed 80
patients, 48 men and 32 women for sexual function and quality of life after
pelvic fractures. Fourteen female patients (43.8%) had sexual dysfunction. They
had a Female Sexual Dysfunction Index of less than 25.5. There was a statistically
significant decrease in all the six assessed domains, namely desire, arousal,
lubrication, orgasm, satisfaction, and pain. Injuries to the urinary tract and
open reduction and internal fixation were statistically significant risk factors
for sexual dysfunction. However, these papers gave no information about how
these patients were managed. Patil et al.11 reported five patients with
dyspareunia, dymenorrhoea, widely separated labia, and urinary incontinence
after a fractured pelvis. A transpubic approach was used for surgical treatment
of these patients. The treatment included excision of the parts of bone that
cause genitourinary obstruction and reconstruction on the urinary tract. Lee et
al.12 reported a female patient with a malunited pelvic fracture causing
dyspareunia that was treated by surgical excision of the bone through an
inguinal approach and excision of the scar tissue through transvaginal approach.
In 1974, Sinha 13 reported a patient with an osteomalacic deformed pelvis
causing obstructive dyspareunia in a 22-year-old woman. Dane et al.14 reported a
patient with postosteomalacic contracted pelvic outlet, with CT showing multiple
pseudo fractures on both ischial rami and looser zones seen on radiographs. Both
patients were treated by excision of the ischial rami by oblique incisions over
the labia majora. In this case, we believed that a direct approach to the
inferior pubic ramus and ischial ramus would be the most suitable and simplest
being an easily planned and performed surgical procedure.8 Owing to the
nontraumatic etiology of her condition, there was no need to do any transvaginal
approaches or to excise any scar tissue. The drawback of the approach we used is
the possibility of pudendal nerve injury in Alcock canal, which we avoided by
strict subperiosteal dissection.

The enlargement of the inferior pubic and ischial rami in this patient can be
understood in light of the better understanding of the actions of the parathyroid
hormone. The elevated levels of parathyroid hormone can cause increased cortical
porosity and thinning of cortical bone.15 However, the effect is very different
on cancellous bone where it improves the bone mass. Various studies have been
run to investigate the mechanism of new bone formation but with no conclusive
evidence.5,16,17 The limitations of the studies mentioned above in explaining
this condition include that they were conducted on patients who received
intermittent parathyroid hormone not patients with primary hyperparathyroidism.
The second thing is that the ischiopubic ramus was not the site of interest in
the studies where biopsies were taken from the spine or iliac crest to monitor
the histological changes due to parathyroid hormone.

In conclusion, this case describes a manifestation of primary hyperparathyroidism,
which has not been previously reported before, namely obstructive dyspareunia
from a deformed pelvis. Our aim in treatment was to remove the cause of the
mechanical obstruction, which was the ischiopubic rami. A simple surgery and
easily performed approach and osteotomy were a successful and practical
solution.

REFERENCES

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5. Compston JE. Skeletal actions of intermittent parathyroid hormone: effects on
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7. Recker RR, Bare SP, Smith SY, et al.. Cancellous and cortical bone architecture
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8. Radley T, Liebig C, Brown J. Resection of the body of the pubic bone, the
superior and inferior pubic rami, the inferior ischial ramus, and the ischial
tuberosity. J Bone Joint Surg Am. 1954; 36-A:855-858. Internet Resources
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9. Vallier HA, Cureton BA, Schubeck D. Pelvic ring injury is associated with
sexual dysfunction in women. J Orthop Trauma. 2012; 26:308-313. Ovid Full Text
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10. Harvey-Kelly KF, Kanakaris NK, Obakponovwe O, et al.. Quality of life and
sexual function after traumatic pelvic fracture. J Orthop Trauma. 2014;
28:28-35. Ovid Full Text Internet Resources Bibliographic Links 

11. Patil U, Nesbitt R, Meyer R. Genitourinary tract injuries due to fracture of
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12. Lee YK, Kim SM, Jeung IC, et al.. Surgical resolution of obstructive
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13. Sinha GP. Partial excision of the ischial rami for post-osteomalacic
contracted pelvic outlet: case report. J Bone Joint Surg Am. 1974; 56:1517-1519.
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14. Dane C, Dane B, Kural C. A rare cause of severe dyspareunia: post-osteomalacic
contracted pelvic outlet. Acta Obstet Gynecol Scand. 2005; 84:407-408. Full Text
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16. Lindsay R, Cosman F, Zhou H, et al.. A novel tetracycline labeling schedule
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