ABSTRACT
BACKGROUND: Although Dega acetabuloplasty is widely used for the treatment of developmental dysplasia of the hip, there is a paucity of data on long-term outcomes. The purpose of the study was to evaluate the rate of residual acetabular dysplasia after Dega acetabuloplasty. METHODS: Patients of a previously reported consecutive series of 35 patients (43 hips) operated by a single surgeon were recontacted for long-term follow-up. Of these, 25 patients (32 hips) consented, with a follow-up rate of 71% (74% of hips). The mean age at the time of surgery was 35 (18 to 65) months. The presence of residual dysplasia was noted according to the lateral center-edge angle of Wiberg, femoral head extrusion index, and Tönnis angle. The latest radiographic outcome was evaluated according to the Severin classification and patients were clinically evaluated according to the modified McKay criteria. RESULTS: The mean follow-up duration of 16.5 (12 to 20) years yielded an average age of 19.2 (14 to 23) years at the time of analysis. According to lateral center-edge angle, femoral head extrusion index, and Tönnis angle, 5 (15.6%) hips were dysplastic and 2 (6.3%) hips were reoperated for resubluxation. Thus, a total of 7 hips (21.9%) were considered to have residual dysplasia. With the exception of 2 hips that underwent further osteotomies, no other hips were re-subluxated or redislocated. Overcoverage was noted in 6 (18.7%) hips. There were 26 Severin group I and II (81.3%), 4 Severin group III (12.5%), and 2 Severin group IV (6.2%) hips. According to modified McKay criteria, 20 (62.5%) hips were excellent, 7 (21.9%) hips were good, and 5 (15.6%) were fair. Severin classification and modified McKay criteria were correlated with dysplasia ( P < 0.05). CONCLUSIONS: Seventy-eight percent of the hips treated by Dega acetabuloplasty for developmental dysplasia of the hip did not have acetabular dysplasia at a mean follow-up of 16 years. Even in well-treated asymptomatic hips, patients should be followed regularly, especially for residual dysplasia. LEVEL OF EVIDENCE: Level IV.
Subject(s)
Acetabuloplasty , Developmental Dysplasia of the Hip , Hip Dislocation, Congenital , Hip Dislocation , Humans , Young Adult , Adult , Child, Preschool , Follow-Up Studies , Developmental Dysplasia of the Hip/diagnostic imaging , Developmental Dysplasia of the Hip/surgery , Retrospective Studies , Radiography , Hip Dislocation, Congenital/diagnostic imaging , Hip Dislocation, Congenital/surgery , Hip Dislocation/diagnostic imaging , Treatment Outcome , Acetabulum/diagnostic imaging , Acetabulum/surgeryABSTRACT
BACKGROUND: Triple pelvic osteotomy (TPO) is indicated when the anatomic and functional realignment of the hip joint is needed. Although the traditional approach for TPO involves a separate incision for ischial cut, there has been a trend for single-incision TPO in recent years. This study aims to compare the clinical and radiologic results of 2 different approaches. METHODS: Forty-two hips of 39 patients treated using TPO with a minimum of 24 months of follow-up were included in our cohort. Demographics, perioperative, and radiologic parameters were evaluated. Harris Hip Score and International Hip Outcome Tool were used for clinical evaluation. RESULTS: A single anterolateral incision approach was used in 18 hips (17 patients), whereas a 3-incision approach was used in 24 hips (22 patients). The mean follow-up was 4.7 years in the 3-incision group and 3.8 years in the single-incision group ( P =0.43), with mean surgery age at 8.7 years (range, 5.4 to 12) for single-incision and 9.7 years (range, 7.7 to 11.7) for 3e-incision ( P =0.22). There were no significant differences observed between the 2 groups concerning radiographic measurements, complications, and functional scores. The mean surgical time was 118.6 minutes in the single-incision group and 97.9 minutes in 3-incision group ( P =0.036). Mean intraoperative blood loss was 181.7 ml in the single-incision group and 243.4 ml in 3-incision group ( P =0.028). Three-incision group demonstrated significantly higher intraoperative blood loss, leading to lower hemoglobin values ( P =0.042). CONCLUSION: The single-incision TPO demonstrated similar outcomes compared with the traditional 3-incision approach in terms of radiologic correction and functional improvement. The single-incision technique exhibited advantages such as reduced intraoperative blood loss and potential benefit of decreased pain due to fewer scars. However, it required a longer surgical time compared with the 3-incision approach. Surgeons should consider patient-specific factors and their expertise when selecting the most appropriate approach for each case. LEVEL OF EVIDENCE: Level III-retrospective comparative series.
Subject(s)
Osteotomy , Pelvic Bones , Humans , Osteotomy/methods , Female , Male , Child , Follow-Up Studies , Pelvic Bones/surgery , Pelvic Bones/diagnostic imaging , Treatment Outcome , Retrospective Studies , Child, Preschool , Hip Joint/surgery , Hip Joint/diagnostic imaging , Operative Time , Radiography/methods , Blood Loss, Surgical/statistics & numerical dataABSTRACT
BACKGROUND: Supracondylar humerus fractures (SHFs) are common pediatric injuries, with type II fractures being a topic of debate regarding optimal treatment. Our goals are to compare the functional and radiographic outcomes of conservative and surgical treatment of type II SHFs and their subgroups and to identify parameters for determining the optimal treatment option. METHODS: We retrospectively reviewed a total of 55 patients (23 conservative, 32 surgical) between 2010 and 2020. The mean follow-up was 66 months. Neurovascular status, range of motion, complications, and functional scores (Mayo elbow, Quick DASH) were evaluated. We performed radiographic assessment on initial, postreduction, and final follow-up radiographs using the humerocondylar angle (HCA), Baumann angle, and anterior humeral line (AHL). RESULTS: Neither groups nor subgroups showed significant differences in clinical and functional outcomes. HCA was significantly higher in the operative group than in the conservative group. The subgroup analysis revealed that the HCA difference resulted from the difference between the conservative IIb and operative IIb subgroups. None of the patients required a corrective osteotomy, but 1 patient initially treated conservatively underwent operative treatment due to loss of reduction. CONCLUSIONS: Reconstructing the sagittal, coronal, and rotational alignment in type II SHFs led to good mid-term results in the range of motion and functional scores for the elbow joint, whether conservative or surgical treatment was used. A limited range of remodeling might be expected in the long term. LEVEL OF EVIDENCE: Level III-retrospective comparative study.
ABSTRACT
BACKGROUND: Tension band plating is widely used in the surgical treatment of coronal plane deformities around the knee. The rebound phenomenon after implant removal is a common complication of this technique. Overcorrection of joint orientation angles is a method to minimize the effect of the rebound phenomenon. This study aims to investigate the natural course of overcorrected joint orientation angles after plate removal in patients with genu valgum deformity. METHODS: Patients who underwent hemiepiphysiodesis with tension band plating due to genu valgum deformity between 2010 and 2019 were retrospectively analyzed. Mechanical lateral distal femoral angles (mLDFA) and mechanical medial proximal tibial angles were calculated before plate application, before implant removal, and at the last follow-up. At the implant removal, mLDFA>90 degrees and mechanical medial proximal tibial angles <85 degrees were accepted as overcorrected. RESULTS: Seventy-two segments from 45 patients were included. For femoral valgus deformities (n=59), the mean mLDFAs at index surgery, implant removal, and the last follow-up were 79.8±3.9 degrees, 95.5±3.7 degrees, and 87.3±5.1 degrees, respectively. In the more and less than 10 degrees rebound groups, the median age of patients at index surgery were 66 and 101 months ( P =0.04), the mLDFA during implant removal were 97.8 degrees and 94.4 degrees ( P =0.005), and the mean amount of correction in mLDFA was 17 degrees and 13 degrees ( P =0.001), respectively. At the last follow-up, joint orientation angles were found to be still overcorrected in 16 (22%), within normal limits in 36 (50%), and undercorrected in 20 (28%) segments. Ten (13%) segments required additional surgery due to residual deformity. CONCLUSIONS: Overcorrection with tension band plating is an effective modality in the treatment of genu valgum deformity. Rebound after plate removal increases as the age at index surgery decreases and the amount of conscious overcorrection increases. Most segments return to normal joint orientation angle limits after overcorrection. We recommend a mean of 5 degrees routine overcorrection in patients with genu valgum deformity to overcome the rebound phenomenon and to make future interventions easier if ever needed. LEVEL OF EVIDENCE: Level III.
Subject(s)
Genu Valgum , Humans , Child, Preschool , Child , Genu Valgum/surgery , Genu Valgum/etiology , Retrospective Studies , Knee Joint/surgery , Knee Joint/abnormalities , Lower Extremity , Knee , Tibia/surgeryABSTRACT
BACKGROUND: Fibular hemimelia (FH) represents the most common deficiency of the long bones and is associated with multiple deformities. Reconstructive treatment with external fixators in FH restores normal lower extremity alignment and length with plantigrade feet for a balanced and effective gait. The aim of this study is to evaluate the outcomes of lower limb lengthening and simultaneous tri-plane deformity correction with a computer-assisted hexagonal external fixator in children with FH. METHODS: A retrospective review was performed for FH cases treated with a computer-assisted hexagonal external fixator in a tertiary referral center. Leg length discrepancy (LLD), interphyseal angles, tibiocalcaneal distances, healing index (HI), and callus shapes were analyzed for radiologic evaluation, and the Pediatric Quality of Life Inventory (PedsQL) was used for functional assessment. Limbs with HI <50 days/cm, PedsQL >75, and without regenerate fractures were considered successful lengthenings. RESULTS: Twenty-four limbs of 23 patients were included. The limbs were lengthened for a mean of 7.24 cm (range, 4.7 to 15.6). The initial LLD of 5.6 cm (range, 0.5 to 19 cm) increased to 1.7 cm (range, 0.1 to 6 cm), and the mean interphyseal angle was 12.7 degree (range, 1.5 to 54.2 degree), tibiocalcaneal distance was 0.85 cm (range, 0.1 to 1.7) at final follow-up. The most common regenerated bone morphology was cylindrical, as seen in 11 limbs (45.8%). The average PedsQL score was 83.5 (range, 69.5 to 96.7). Sixteen limbs (66.7%) had successful lengthening at their first, and 4 limbs (80%) had successful lengthening at their second surgeries. Seven limbs had complications requiring surgical intervention (29.1%), with 3 (12.5%) regenerate fractures after external fixators removal. CONCLUSIONS: Limb reconstruction with computer-assisted hexapod fixators is a successful and reliable option for the treatment of LLD in FH, and patients demonstrate good functional outcomes. Surgeons should be aware of potential complications and should utilize prophylactic measures when necessary. LEVELS OF EVIDENCE: Level III, retrospective comparative study.
Subject(s)
Bone Lengthening , Ectromelia , Fractures, Bone , Child , Humans , Ectromelia/diagnostic imaging , Ectromelia/surgery , Ectromelia/complications , Retrospective Studies , Quality of Life , Bone Lengthening/adverse effects , External Fixators/adverse effects , Leg Length Inequality/etiology , Lower Extremity , Fractures, Bone/etiology , Computers , Treatment Outcome , Tibia/abnormalitiesABSTRACT
BACKGROUND: The present study aims to investigate the frequency of recurrence and tethering effect after only metaphyseal screw removal (sleeper plate technique) compared with the conventional complete plate removal in the treatment of lower extremity deformities with guided growth surgery. METHODS: Seventy-two patients (107 limbs) treated by an 8-plate hemiepiphysiodesis technique around the knee joint were evaluated. After the desired correction, only metaphyseal screw was removed (sleeper plate group) in 35 limbs (25 patients), whereas both screws and plate were removed (plate removal group) in 72 limbs (47 patients). An increase of 5 degrees or more in joint orientation angles in the direction of the initial deformity was considered as recurrence. The rate of rebound, tethering, and maintenance of correction in groups was analyzed at the latest follow-up (mean of 49 mo). RESULTS: The mean age of the patients was 97 months (range: 80 to 129 mo) at the time of index surgery. After a mean of 49 months (range: 16 to 86), 17 (48.5%) limbs maintained the desired stable correction in the sleeper plate group compared with 59 stable limbs (72.2%) in the plate removal group ( P <0.001). There was no statistically significant difference regarding recurrence between the sleeper plate group and the plate removal group (34.3% vs. 27.8%, respectively) ( P =0.216). Reinsertion of the metaphyseal screw was possible 8/12 limbs, and the remaining 4 limbs underwent further surgeries. There were 6 limbs (17.3%) of tethering in the sleeper plate group, and 4/6 limbs required further corrective surgeries. The remaining 2 limbs with slight tethering did not require further surgeries. CONCLUSIONS: Removing only metaphyseal screw increases the risk of tethering. In addition, reinsertion of the screw may not be possible in all cases due to bony growth, and further corrective surgeries may be necessary. Close follow-up is required if the sleeper plate technique is to be applied. LEVEL OF EVIDENCE: Level III.
Subject(s)
Knee Joint , Plastic Surgery Procedures , Humans , Child , Knee Joint/surgery , Knee Joint/abnormalities , Arthrodesis/adverse effects , Extremities , Postoperative Complications/etiology , Bone Plates/adverse effects , Retrospective StudiesABSTRACT
BACKGROUND AND AIM: Legg-Calve-Perthes disease (LCPD) is still an enigma. Hemostatic abnormalities have been indicated in the pathogenesis. We had previously demonstrated enhanced tissue factor pathway inhibitor response, increased global fibrinolytic capacity, and an increase in thrombomodulin in patients with LCPD compared with healthy individuals. These studies emphasized the role of vascular endothelium in pathogenesis of the LCPD. P-selectin is expressed on activated platelets and endothelial cells, and E-selectin is expressed on activated endothelial cells. The aim of this study was to assess circulating E-selectin and P-selectin levels in LCPD patients, which might reflect an endothelium activation and/or injury. MATERIALS AND METHODS: The study included 85 pediatric patients. Group I consisted of 55 patients with LCPD and group II (control) consisted of 30 healthy children. Peripheral venous blood concentrations of E-selectin and P-selectin levels were measured with a commercially available assay. RESULTS: Mean age was 8.41±2.73 years in group I and 8.83±2.92 years in group II. Both E-selectin and P-selectin levels were higher in LCPD patients in comparison with the age-matched controls. E-selectin was 54.92±18.84 pg/mL in group I, 45.54±15.31 pg/mL in group II and P-selectin was 46.40±20.35 pg/mL in group I, 36.92±9.84 pg/mL in group II (P=0.022 and P=0.019, respectively). CONCLUSIONS: On the basis of our results, two important endothelium and platelet markers, E-selectin and P-selectin, are upregulated in LCPD. Our results suggested that activated platelets and possibly endothelial activation, as reflected by enhanced P-selectin/E-selectin kinetics, might contribute to the microvascular thrombosis and/or inflammation of LCPD.
Subject(s)
E-Selectin/blood , Endothelium, Vascular/metabolism , Legg-Calve-Perthes Disease/metabolism , P-Selectin/blood , Platelet Activation/physiology , Adolescent , Biomarkers/blood , Blood Platelets/metabolism , Child , Child, Preschool , Female , Humans , Male , Microvessels/metabolism , Solubility , Thrombosis/metabolism , Vasculitis/metabolismABSTRACT
OBJECTIVES: The aim of this study is to investigate whether rigid fixation after triple pelvic osteotomy (TPO) utilizing a 3.5-mm locking plate and screws without hip spica cast can provide enough stability and prevent correction loss in pediatric patients with developmental dysplasia of the hip (DDH) and Legg-Calvé-Perthes disease (LCPD). PATIENTS AND METHODS: A total of 21 hips of 21 pediatric patients (9 males, 12 females; mean age: 9.3±2.0 years; range, 6 to 14 years) who underwent rigid fixation with locking plate/screws after TPO for DDH and LCPD between June 2015 and October 2018 were retrospectively analyzed. Preoperative, immediate postoperative, and six-month follow-up anteroposterior radiographs were compared for the Wiberg's center-edge angle (CE), Sharp angle, acetabular coverage of the femoral head (ACFH), and center-head distance discrepancy (CHDD). The patient demographics, surgery time, perioperative complications were evaluated. RESULTS: Underlying diagnosis were DDH in 14 patients and LCPD in seven patients. In patients with DDH, postoperative evaluation showed significant increase in the mean CE angle (5.6±16.1° vs. 30.5±9.3°, respectively) and ACFH (46.4±16.8% vs. 84.5±12.1%, respectively), and a significant decrease in the mean Sharp angle (55.3±6.2° vs. 35.6±7.8°, respectively) and CHDD (14.6±10.7% vs. 6.2±5.6%, respectively). The final follow-up revealed that there was no correction loss in these parameters. In the patients with LCPD, postoperative evaluation showed a significant increase in the mean CE (20.1±11.1° vs. 38.3±9.6°, respectively) and ACFH (62.9±18% vs. 91.4±10.1%, respectively), and a significant decrease in the mean Sharp angle (46±3.6° vs. 25.2±5.5°, respectively). The final follow-up revealed that there was no correction loss in radiological parameters. No perioperative complications were noted. CONCLUSION: Our study results suggest that rigid fixation construct with a 3.5-mm locking plate and screws without hip spica cast can provide adequate stability to allow early mobilization following TPO in children without any loss of correction, until bony healing at the osteotomy sites.
Subject(s)
Bone Plates/statistics & numerical data , Bone Screws/statistics & numerical data , Casts, Surgical/statistics & numerical data , Hip Dislocation/surgery , Legg-Calve-Perthes Disease/surgery , Osteotomy/statistics & numerical data , Pelvic Bones/surgery , Adolescent , Child , Female , Humans , Male , Retrospective Studies , Treatment Outcome , TurkeyABSTRACT
Although osteochondral autograft transplantation (OAT) in the knee and ankle has gained much popularity on the other hand the technique has rarely been applied in the femoral head. In this article, we present a 15-year-old female patient with unstable chondral lesion on the right femoral head. She had a history of open reduction for developmental dysplasia which resulted in avascular necrosis. She had coxa magna, breva and trochanteric overgrowth along with an unstable chondral lesion on the superolateral part of the femoral head. She underwent OAT for chondral lesion, femoral head reshaping and relative neck lengthening. Her Harris Hip Score improved from 55 to 90 in the 18 months of follow-up. The case is unique in a way that the autografts were harvested from the anterior part of the same femoral head that was already intended to be removed during osteochondroplasty.
Subject(s)
Bone Transplantation , Cartilage, Articular/transplantation , Femur Head/surgery , Femur Neck/surgery , Hip Dislocation, Congenital/surgery , Adolescent , Female , Hip Dislocation, Congenital/physiopathology , Hip Joint/physiopathology , Humans , Transplantation, Autologous/methodsABSTRACT
A stump neuroma is an attempt for the repair of a nerve following amputation. This article presents a case of a 60-year-old woman who was suspected of having a local recurrence on the chest wall following amputation of the left arm for a malignant mesenchymal tumor. The tumor did not show any invasion to adjacent structures thereby any necessity for chest wall resection and reconstruction. The patient underwent local excision of the tumor with reamputation of the branches of the lower brachial plexus, subclavian artery, and proximal one third of the clavicle. The tumor was eventually diagnosed as a brachial plexus neuroma. Although rare, cases that require left pneumonectomy with a transthoracic forequarter amputation including brachial plexus resection have been reported. Transection of the brachial plexus also may be needed during resection of a superior sulcus tumor. These cases often undergo an aggressive chest wall resection that includes the subclavian artery and vein in addition to the brachial plexus, particularly in tumors involving the lower truncus. Therefore, when recurrent chest wall tumor is suspected in such cases, stump neuroma resulting from transection of the brachial plexus should be included in the differential diagnosis.
Subject(s)
Brachial Plexus Neuropathies/diagnosis , Brachial Plexus Neuropathies/surgery , Neuroma/diagnosis , Neuroma/surgery , Peripheral Nervous System Neoplasms/diagnosis , Peripheral Nervous System Neoplasms/surgery , Chondrosarcoma, Mesenchymal/diagnosis , Diagnosis, Differential , Female , Humans , Middle Aged , Neoplasm Recurrence, Local/diagnosis , Thoracic Neoplasms/diagnosis , Treatment OutcomeABSTRACT
Whether acetabular volume increases or decreases after acetabular Dega osteotomy is not known. The purpose of this study is to determine the effect of Dega osteotomy on the volume of the acetabulum in patients with developmental dysplasia of the hip. Nine hips of seven patients with developmental dysplasia of the hip that have undergone Dega osteotomy were included in the study. The acetabular index, acetabular depth, and acetabular volume of each hip were calculated before and after surgery. Magnetic resonance imaging was used for the measurement of the acetabular volume. The difference between the preoperative and postoperative values of acetabular index, acetabular depth, and acetabular volume was statistically significant. We conclude that Dega acetabular osteotomy increases the volume of the acetabulum.
Subject(s)
Acetabulum/pathology , Hip Dislocation, Congenital/surgery , Osteotomy/methods , Plastic Surgery Procedures/methods , Child, Preschool , Female , Hip Dislocation, Congenital/pathology , Humans , Infant , Magnetic Resonance Imaging/instrumentation , Magnetic Resonance Imaging/methods , MaleABSTRACT
BACKGROUND: Legg-Calvé-Perthes disease (LCPD) is the idiopathic avascular necrosis of the femoral head in childhood. The pathologic changes seen in the femoral head are likely a result of vascular factors. Blood neutrophil to lymphocyte (N/L) ratio is a simple marker of subclinical inflammation. This study aims to to analyse the predictive ability of N/L ratio for the prognosis in LCPD patients. METHODS: Patients who had been diagnosed as LCPD from 2008 to 2014 were investigated retrospectively and 40 LCPD patients (33 male and 7 female) and 25 healthy age and sex-matched children (controls) were included in the study. LCPD patients were divided into 2 groups according to expected prognosis (good prognosis expected Herring A and B patients as Group I and poor prognosis expected Herring B-C and C patients as Group II) and healthy children (control) were included in Group III. All the patients' hematological markers were analysed. RESULTS: Mean age was 7.1 ± 2.0 years in group I (4.9-12 years), 8.3 ± 2.2 years (4-12.5 years) in group II and 7.8 ± 1.3 years (6-12 years) in group III. Mean values for Groups I, II and III for neutrophil to lymphocyte (N/L) ratio were 1.13 ± 0.65, 1.75 ± 0.95, 1.08 ± 0.37, respectively. The mean neutrophil to lymphocyte (N/L) ratio of Group II was higher than the other 2 groups (p = 0.003). CONCLUSIONS: N/L ratio may give us information about the natural course of LCPD and may be used as independent predictor of prognosis in patients with LCPD.
Subject(s)
Legg-Calve-Perthes Disease/blood , Legg-Calve-Perthes Disease/diagnosis , Lymphocytes , Neutrophils , Child , Child, Preschool , Female , Humans , Leukocyte Count , Male , Predictive Value of Tests , Prognosis , Retrospective StudiesABSTRACT
Nonunion and major complications are not very common in pediatric fractures. No commonly accepted methods exist for the treatment of these fractures. In this study, treatment of complicated pediatric femoral fractures with titanium elastic nails was evaluated. Five girls and four boys with nine femoral fractures constituted the group. The mean age of the patients was 8.7 (5-11) years and the mean follow-up was 46.6 (24-84) months. Five of them had implant failure and nonunion, two of them had loss of position in the external fixator, one of them had incomplete union and loss of position in the external fixator and the other had femoral fracture in the lengthening segment of the femur and had loss of position and incomplete union in the spica cast. All the patients were treated with a closed or open reduction of the fracture, debridement if treated with open method and fixation of the fracture with titanium elastic nails. Complete union was achieved in all the patients within the 6-9 months. No neurovascular injury or infection was reported in the group. The result of the treatment showed that fixation of complicated pediatric femoral fractures with intramedullary titanium elastic nails is a good option.
Subject(s)
Bone Nails , Femoral Fractures/surgery , Fracture Fixation, Intramedullary/instrumentation , Fracture Fixation, Intramedullary/methods , Child , Child, Preschool , Debridement , Female , Follow-Up Studies , Fracture Healing , Fractures, Ununited/surgery , Humans , Internal Fixators , Male , Postoperative Complications , Prosthesis Design , Retrospective Studies , TitaniumABSTRACT
The precise pathogenetic basis of Legg-Calve-Perthes disease (LCPD) is currently unknown. Hemostatic abnormalities, i.e., hypercoagulability and/or hypofibrinolysis, were proposed in the genesis of the LCPD. Deficiency of tissue factor pathway inhibitor (TFPI), a critical natural anticoagulant molecule, may lead to a prothrombotic state in a wide variety of conditions. The aim of this study is to assess the circulating TFPI pool in the LCPD. Group I consisted of 44 patients with LCPD and group II comprised 38 healthy children. Median (IQR) TPFI concentration was significantly higher in the group I (p < .0001). Enhanced TFPI response could be regarded as a compensatory defense mechanism against ongoing local microvascular events of occlusion and revascularization of LCPD. TFPI molecule may be an important link between the crossroads of the LCPD genesis and pathogenetic microvascular changes in the disease course. Further investigations are needed to shed light on the endothelial anticoagulant kinetics, the unique microvascular compromise, and the self-limiting nature of the disease.