Functional and biochemical improvement following total knee arthroplasty in early postoperative period.

OBJECTIVE: There are very few studies about total knee arthroplasty biomechanical and biochemical effects in the early postoperative period. The aim of this study was to investigate the effect of total knee arthroplasty on pain intensity, knee joint valgus angle, malalignment, functional status, knee joint position sense, and cytokine levels. METHODS: A total of 29 patients (female/male: 24/5) who underwent total knee arthroplasty were included in the late-stage knee osteoarthritis group, and 22 patients (female/male: 13/9) with grade 4 osteoarthritis were included in the early-stage knee osteoarthritis group. The visual analog scale and the Western Ontario and McMaster Universities Osteoarthritis Index were used to evaluate the pain intensity and functional status. Alignment and knee position sense measurements were also calculated. Systemic venous blood samples were taken to evaluate the interleukin-6, tumor necrosis factor-alpha, and interleukin-1 beta cytokine levels. RESULTS: In the study group, there were positive improvements in pain intensity, functional status, valgus angle, malalignment, amount of joint position sense deviation at 70° knee flexion angle parameters, and interleukin-6 of patients at the postoperative 6th week compared to the preoperative period (p<0.05). The patients in the study group had similar or better results in pain intensity, functional status, valgus angle, malalignment, amount of joint position sense deviation at 35°, 55°, and 70° knee flexion angles parameters, and in interleukin-6, compared to the control group at postoperative 6th week. CONCLUSION: Total knee arthroplasty provides improvements in pain, function, valgus angle, joint position sense, and interleukin-6 in the early postoperative period.

Tibial slope, remnant preservation, and graft size are the most important factors affecting graft healing after ACL reconstruction.

PURPOSE: The aim of this study was to determine the anatomic, operative and biological factors that influenced graft healing after single-bundle anterior cruciate ligament (ACL) reconstruction. METHODS: One hundred fourteen consecutive patients who underwent anatomic single-bundle ACL reconstruction with quadrupled hamstring tendon autografts between 2016 and 2019 were retrospectively analyzed. Ninety-four patients met the inclusion criteria with minimum follow-up of 12 months. Patients were evaluated with multiple clinical measurements, including International Knee Documentation Committee Subjective Knee Form (IKDC-SKF), Lyshom Scores, and Marx activity scale. To evaluate graft healing, the signal-to-noise quotient (SNQ) was measured at intra-articular graft and intra-tunnel integration were evaluated on magnetic resonance imaging (MRI) at one year after surgery. Potential factors affecting graft healing, including age, sex, body mass index, time from injury to surgery, posterior tibial slope, lateral femoral condyle ratio, notch width index, meniscal injury, remnant preservation, tunnel aperture locations, graft size, graft bending angle, graft/remaining notch volume ratio were evaluated for their association with graft SNQ value by stepwise regression analysis. RESULTS: A total of 94 patients were evaluated with mean follow-up 28.5 ± 9 months. Univariate regression analysis showed that posterior tibial slope, notch width index, remnant preserving procedure, high femoral tunnel, anterior tibial tunnel, graft bending angle, and graft/remaining notch volume ratio significantly associated with graft SNQ values. Multivariate regression analysis showed that lateral tibial slope, remnant preservation, and graft/remaining notch volume ratio were independent factors correlated with graft SNQ values. Also, the graft SNQ values was weakly correlated with femoral tunnel integration and Marx activity scale at one year. There was no correlation between graft SNQ values and IKDC-SKF and Lysholm scores. There was no correlation between graft SNQ values and International Knee Documentation Committee and Lysholm scores. CONCLUSIONS: Tibial slope, remnant preservation and graft/remaining notch volume ratio were significant independent associated factors of graft SNQ value at one year. The graft SNQ values were also weakly correlated with femoral tunnel integration and the Marx activity scale. These factors should be taken into account for ensuring the ideal graft healing and for the return to sport decision-making. LEVEL OF EVIDENCE: Level IV.

Functional and biochemical improvement following total knee arthroplasty in early postoperative period

SUMMARY OBJECTIVE: There are very few studies about total knee arthroplasty biomechanical and biochemical effects in the early postoperative period. The aim of this study was to investigate the effect of total knee arthroplasty on pain intensity, knee joint valgus angle, malalignment, functional status, knee joint position sense, and cytokine levels. METHODS: A total of 29 patients (female/male: 24/5) who underwent total knee arthroplasty were included in the late-stage knee osteoarthritis group, and 22 patients (female/male: 13/9) with grade 4 osteoarthritis were included in the early-stage knee osteoarthritis group. The visual analog scale and the Western Ontario and McMaster Universities Osteoarthritis Index were used to evaluate the pain intensity and functional status. Alignment and knee position sense measurements were also calculated. Systemic venous blood samples were taken to evaluate the interleukin-6, tumor necrosis factor-alpha, and interleukin-1 beta cytokine levels. RESULTS: In the study group, there were positive improvements in pain intensity, functional status, valgus angle, malalignment, amount of joint position sense deviation at 70° knee flexion angle parameters, and interleukin-6 of patients at the postoperative 6th week compared to the preoperative period (p<0.05). The patients in the study group had similar or better results in pain intensity, functional status, valgus angle, malalignment, amount of joint position sense deviation at 35°, 55°, and 70° knee flexion angles parameters, and in interleukin-6, compared to the control group at postoperative 6th week. CONCLUSION: Total knee arthroplasty provides improvements in pain, function, valgus angle, joint position sense, and interleukin-6 in the early postoperative period.

Effectiveness and reliability of traditional open surgery in atypical localizations of osteoid osteoma.

OBJECTIVES: This study aims to evaluate traditional open surgery results of osteoid osteomas (OOs) in atypical localizations and explore whether open surgery can be a safe alternative in localizations where radiofrequency ablation (RFA) may not be suitable. PATIENTS AND METHODS: A total of 26 patients (20 males, 6 females; mean age 23.3±14.2 years; range, 4 to 65 years) having OO in atypical localizations between January 2008 and January 2017 were retrospectively evaluated. All patients underwent traditional open surgery under anesthesia. All patients were followed-up, and their clinical success and complications were evaluated. The pain intensity was assessed using the visual analog scale (VAS). RESULTS: The postoperative first year VAS score was 0.2±0.5 (range, 0-2) (p<0.005). None of the patients showed any complications. No recurrences were reported at the end of the first year. All patients were relieved from pain due to OO in atypical localizations. CONCLUSION: Although RFA has been accepted as the preferred treatment for OO, traditional open surgery may still be used for OO in atypical localizations yielding perfect clinical results with fewer complications. It is a safe alternative for localizations such as carpal bones, distal ulna, hand and foot where RFA complications can be seen more frequently. In these localizations, traditional open surgery can be recommended as the first treatment choice.

A Comparison of the Effects of Grayanotoxin-Containing Honey (Mad Honey), Normal Honey, and Propolis on Fracture Healing.

OBJECTIVES: Delayed healing and non-union of fractures have a significant effect upon patient morbidity. Studies have therefore largely concentrated on accelerating fracture healing. This study was intended to compare the effect of "mad honey" and propolis on fracture healing using radiological and histopathological analysis. SUBJECTS AND METHODS: Femur fracture was surgically performed on 48 rats, followed by fixation. Animals were then divided into 8 groups: 2 control groups (15- and 30-day) and 6 treatment groups (15- and 30-day normal honey, 15- and 30-day "mad honey," and 15- and 30-day propolis). Rats were sacrificed at the end of these periods, and radiological and histological examinations were performed. RESULTS: Radiological healing in the propolis group after 15-day therapy was statistically better than in the control (p = 0.004) and normal honey (p = 0.006) groups. After 30-day therapy, healing in the propolis group (p = 0.005) and grayanotoxin-containing "mad honey" group (p = 0.007) were significantly better than in the control group. Histologically, there was a statistically significant difference between the 15-day propolis group and the other groups (control, honey, mad honey: p = 0.003, p = 0.003, and p = 0.002, respectively). We also found a statistically significant difference when the 30-day propolis group (p = 0.005) and "mad honey" group (p = 0.007) were compared to the control group. CONCLUSIONS: This study shows that grayanotoxin-containing "mad honey" and propolis can accelerate fracture healing.

Ginkgo Biloba improves bone formation during fracture healing: an experimental study in rats.

OBJECTIVES: Ginkgo biloba extract (EGb 761) is a plant extract obtained from the leaves of the G. biloba tree. The aim of this study was to assess the histological and radiological effects of G. biloba extract on fracture healing in an experimental fracture model using rat femurs. METHODS: Forty-eight female Sprague-Dawley rats (weight: 195-252 g; age: 20 weeks) were used in the study. The rats were randomly divided into six groups (n=8). A transverse fracture was made in the middle of the right femur of each rat and fixed with a Kirschner wire. The G. biloba groups received 60 mg/kg oral G. biloba extract once daily. No medication was given to the control groups. On days 7, 21 and 35, both sets of femurs were evaluated radiologically and histopathologically. RESULTS: Histological evaluation revealed that the G. biloba groups had significant differences at 21 and 35 days (p<0.05). The G. biloba group showed a significant difference in terms of bone formation on day 21 when compared to the control group (p<0.05). CONCLUSIONS: This study indicated that the use of G. biloba extract accelerated fracture healing. Both radiological and histological differences were detected, but the histological differences were more remarkable. Level of Evidence I, High Quality Randomized Trial.

OBJETIVOS: O extrato de Ginkgo biloba (EGb 761) é um extrato vegetal obtido das folhas da árvore Ginkgo biloba. O objetivo deste estudo foi avaliar os efeitos histológicos e radiológicos do extrato de Ginkgo biloba sobre a consolidação de fraturas em um modelo experimental de fratura em fêmures de rato. MÉTODOS: Foram utilizados 48 ratos Sprague-Dawley fêmeas (peso: 195-252 g, idade: 20 semanas). Os ratos foram divididos randomicamente em seis grupos (n = 8). Uma fratura transversal foi feita no meio do fêmur direito de cada rato e fixada com fio de Kirschner. Os grupos G. biloba receberam 60 mg/kg de G. biloba por via oral uma vez por dia. Não foi administrada nenhuma medicação aos grupos de controle. Nos dias 7, 21 e 35, ambos os fêmures foram avaliados radiológica e histopatologicamente. RESULTADOS: A avaliação histológica revelou que os grupos G. biloba apresentaram diferenças significativas aos 21 e 35 dias (p < 0,05). O grupo G. biloba mostrou uma diferença significativa em termos de formação óssea no dia 21 quando comparado com o grupo controle (p < 0,05). CONCLUSÕES: Este estudo indicou que o uso de extrato de G. biloba acelerou a consolidação de fraturas. As diferenças radiológicas e histológicas foram detectadas, mas as diferenças histológicas foram mais notáveis. Nível de Evidência I, Estudo Clínico Randomizado de Alta Qualidade.

Ginkgo Biloba improves bone formation during fracture healing: an experimental study in rats / Ginkgo Biloba melhora a formação dos ossos durante a consolidação da fratura: estudo em ratos

ABSTRACT OBJECTIVES: Ginkgo biloba extract (EGb 761) is a plant extract obtained from the leaves of the G. biloba tree. The aim of this study was to assess the histological and radiological effects of G. biloba extract on fracture healing in an experimental fracture model using rat femurs. METHODS: Forty-eight female Sprague-Dawley rats (weight: 195-252 g; age: 20 weeks) were used in the study. The rats were randomly divided into six groups (n=8). A transverse fracture was made in the middle of the right femur of each rat and fixed with a Kirschner wire. The G. biloba groups received 60 mg/kg oral G. biloba extract once daily. No medication was given to the control groups. On days 7, 21 and 35, both sets of femurs were evaluated radiologically and histopathologically. RESULTS: Histological evaluation revealed that the G. biloba groups had significant differences at 21 and 35 days (p<0.05). The G. biloba group showed a significant difference in terms of bone formation on day 21 when compared to the control group (p<0.05). CONCLUSIONS: This study indicated that the use of G. biloba extract accelerated fracture healing. Both radiological and histological differences were detected, but the histological differences were more remarkable. Level of Evidence I, High Quality Randomized Trial.

RESUMO OBJETIVOS: O extrato de Ginkgo biloba (EGb 761) é um extrato vegetal obtido das folhas da árvore Ginkgo biloba. O objetivo deste estudo foi avaliar os efeitos histológicos e radiológicos do extrato de Ginkgo biloba sobre a consolidação de fraturas em um modelo experimental de fratura em fêmures de rato. MÉTODOS: Foram utilizados 48 ratos Sprague-Dawley fêmeas (peso: 195-252 g, idade: 20 semanas). Os ratos foram divididos randomicamente em seis grupos (n = 8). Uma fratura transversal foi feita no meio do fêmur direito de cada rato e fixada com fio de Kirschner. Os grupos G. biloba receberam 60 mg/kg de G. biloba por via oral uma vez por dia. Não foi administrada nenhuma medicação aos grupos de controle. Nos dias 7, 21 e 35, ambos os fêmures foram avaliados radiológica e histopatologicamente. RESULTADOS: A avaliação histológica revelou que os grupos G. biloba apresentaram diferenças significativas aos 21 e 35 dias (p < 0,05). O grupo G. biloba mostrou uma diferença significativa em termos de formação óssea no dia 21 quando comparado com o grupo controle (p < 0,05). CONCLUSÕES: Este estudo indicou que o uso de extrato de G. biloba acelerou a consolidação de fraturas. As diferenças radiológicas e histológicas foram detectadas, mas as diferenças histológicas foram mais notáveis. Nível de Evidência I, Estudo Clínico Randomizado de Alta Qualidade.

Prospective Randomized Comparison of the Effectiveness of Radiation Therapy and Local Steroid Injection for the Treatment of Plantar Fasciitis.

PURPOSE: The purpose of this study was to conduct a randomized trial of radiation therapy for plantar fasciitis and to compare radiation therapy with local steroid injections. METHODS AND MATERIALS: Between March 2013 and April 2014, 128 patients with plantar fasciitis were randomized to receive radiation therapy (total dose of 6.0 Gy applied in 6 fractions of 1.0 Gy three times a week) or local corticosteroid injections a 1 ml injection of 40 mg methylprednisolone and 0.5 ml 1% lidocaine under the guidance of palpation. The results were measured using a visual analog scale, a modified von Pannewitz scale, and a 5-level function score. The fundamental phase of the study was 3 months, with a follow-up period of up to 6 months. RESULTS: The median follow-up period for all patients was 12.5 months (range, 6.5-18.6 months). For the radiation therapy patients, the median follow-up period was 13 months (range, 6.5-18.5 months), whereas in the palpation-guided (PG) steroid injection arm, it was 12.1 months (range, 6.5-18.6 months). After 3 months, results in the radiation therapy arm were significantly superior to those in the PG steroid injection arm (visual analog scale, P<.001; modified von Pannewitz scale, P<.001; 5-level function score, P<.001). Requirements for a second treatment did not significantly differ between the 2 groups, but the time interval for the second treatment was significantly shorter in the PG steroid injection group (P=.045). CONCLUSION: This study confirms the superior analgesic effect of radiation therapy compared to mean PG steroid injection on plantar fasciitis for at least 6 months after treatment.

Giant cell tumor of the metacarpal bone in children: free osteoarticular metatarsal transfer: case report.

We present a successful total resection of metacarpal bone and nonvascularized joint transfer in a giant cell tumor of the fourth metacarpal bone in a 13-year-old girl. At the 6-year follow-up, a good functional outcome was achieved, with 85° range of motion of the metacarpophalangeal joint and no clinical or radiographic evidence of tumor recurrence and no signs of degeneration of the joint.

Effects of human amniotic fluid on fracture healing in rat tibia.

BACKGROUND: Human amniotic fluid (HAF), including hyaluronic acid (HA) and several growth factors, has been used experimentally in tendon, nerve, and cartilage regeneration and in bone defects because of its positive stimulating effects on regeneration potential. This study was performed to investigate whether HAF was effective on fracture healing. MATERIALS AND METHODS: We created 36 tibial fractures in 20-week-old Wistar rats that were divided into three groups. In group 1, fracture lines were instilled with HAF collected at 18th week of the gestation and, in group 2, fracture lines were instilled with HAF obtained at the end of the gestation. HAF which was collected from different period of gestation was used, because the concentration of HA and growth factors in HAF varies considerably during gestation. Group 3 was used as an operative control group. RESULTS: Fracture-healing score was highest in group 1 radiologically at the 3rd and 5th week (P = 0.037, P = 0.018, respectively). In the scintigraphic evaluation, metabolic activity at the fracture site was observed in group 1 more than the others at the 3rd week (P = 0.010). Histologically, the highest scores were obtained from group 1 as compared to other groups at the 3rd and 5th week. In the 5th week, predominant cartilage with some woven bone was observed in group 3, while predominantly woven bone with some cartilage was observed in group 1 (P = 0.036). CONCLUSIONS: Our data suggest that HAF had a positive effect on fracture healing in rat tibia, and also this positive effect was observed more in group 1.

Intraarticular fibroma of the tendon sheath arising from the infrapatellar fat pad in the knee joint.

We report a rare case of fibroma of the tendon sheath arising from the fat pad in the knee. To our knowledge, only four previous cases of fibroma of the tendon sheath in the knee have been reported in the English literature. In our case, magnetic resonance imaging showed low signal intensity in the mass on T1- and T2-weighted images. The mass was excised by mini arthrotomy.

Synovial chondromatosis of the subtalar joint: a case report and review of the literature.

Synovial chondromatosis is an uncommon disorder characterized by the formation of multiple cartilaginous nodules within the synovium and most commonly affects large joints, such as the knee and hip. Diagnosis in synovial chondromatosis is generally confirmed by histology after clinical and radiologic examination. Diagnosis may sometimes be difficult because synovial chondromatosis resembles a soft-tissue mass and may give no radiologic findings. We describe a case of synovial chondromatosis stemming from the subtalar joint, in which diagnosis was difficult clinically and radiologically. The patient presented with pain in the ankle and with a soft-tissue mass. This case is presented with a review of the literature on subtalar joint involvement of synovial chondromatosis.

[Anterior cruciate ligament reconstruction with the peroneus longus tendon]. / Peroneus longus tendonu ile ön çapraz bag rekonstrüksiyonu.

OBJECTIVES: The aim of the study was to evaluate the results of anterior cruciate ligament (ACL) reconstruction using a peroneus longus tendon (PLT) autograft. METHODS: The study included 29 patients (27 males, 2 females; mean age 30 years; range 21 to 39 years) who underwent ACL reconstruction using a PLT autograft and interference nail fixation. Fourteen patients (48.3%) also had partial meniscectomy during surgery. The results were assessed according to the Lysholm scores and the International Knee Documentation Committee (IKDC) scale at the end of at least five years of follow-up. RESULTS: According to the IKDC scale, 17 patients (58.6%) were rated as normal or nearly normal, and 12 patients (41.4%) were rated as abnormal or severely abnormal. The mean Lysholm score was 83.7 (range 45 to 100), with excellent or good results in 23 patients (79.3%). Radiographic examination showed mild (n=10) or moderate (n=1) degenerative changes in the knee joint. Compared with the normal side, no flexion or extension losses occurred in the affected knees. Stability of the ACL was assessed by the Lachman test, which showed normal findings in 12 patients (41.4%), while nine patients had 1+, five patients had 2+, and three patients had 3+ anteroposterior laxity. Pivot-shift test was negative in 13 patients (44.8%); ten patients had 1+ pivot glide, and six patients had 2+ pivot shift. Two patients (6.9%) complained of mild to moderate pressure pain, paresthesia and dysesthesia at the donor site of PLT. No patient experienced ankle joint dysfunction or difficulty in sports activities due to PLT graft transfer. CONCLUSION: Our data show that PLT can be an appropriate autograft source for ACL reconstruction, avoiding potential complications of autografts obtained from the knee region.

Bilateral non-traumatic acetabular and femoral neck fractures due to pregnancy-associated osteoporosis.

Pregnancy-associated osteoporosis is a rare disorder and its pathophysiology remains unknown. We report a case of pregnancy-associated osteoporosis in a 27-year-old primiparous patient who revealed bilateral hip pain during early postnatal period. The plain radiographs and computerized tomography showed bilateral femoral neck and acetabular fractures. The diagnosis of osteoporosis was established by bone mineral density. Diagnostic work-up excluded a secondary osteoporosis. The case was treated successfully by bilateral cementless total hip arthroplasty. Bone mineral density increased after 2 years of treatment with calcium-vitamin D, calcitriol and alendronate. Diagnosis of pregnancy-associated osteoporosis should be suspected when hip pain occurs during pregnancy or in the post-partum period as it can lead to acetabular and femoral neck fractures.

Osteoid osteoma with a multicentric nidus: a case report and review of the literature.

To date, 23 cases with osteoid osteoma (OO) including multiple nidi in single bone have been reported in the world literature. A case report of an 18-year-old boy with an OO on his left femoral neck, which contained double nidi is presented. Plain radiography, computed tomography (CT) and magnetic resonance imaging (MRI) of the proximal femur showed OO with a multicentric nidus. Bone scintigraphy demonstrated increased activity in the left femoral neck region. The tumor was removed with curettage and shaving using lateral approach. The patient was asymptomatic for 5 years after surgery.

Ipsilateral dislocation of the shoulder and elbow joints with contralateral comminuted humeral fracture.

Ipsilateral dislocation of the shoulder and elbow joints is a rare and complex injury. During the last 25 years, only 3 cases have been reported in the literature. We report a 50-year-old woman who suffered ipsilateral elbow and shoulder dislocation with contralateral comminuted humeral fracture. Both shoulder and elbow joints were reduced, but the elbow was dislocated subsequently at follow-up. The reduction in the elbow was stabilized by a Kirschner wire that was removed at 3 weeks, and the elbow was then stable. A U-shaped coaptation splint was applied for the contralateral comminuted humeral fracture. At 6 months, she had acquired a nearly full range of motion of both shoulder and elbow with complete healing in the contralateral humerus. Although rare and complex, ipsilateral shoulder and elbow dislocation, which is the result of a high-energy trauma, can be treated conservatively.

Hip arthroplasty for ochronosis.

Alkaptonuria is a metabolic disorder in which homogentisic acid oxidase is absent. Therefore, homogentisic acid accumulates in cartilage and connective tissues. We can diagnose ochronotic arthropathy, a manifestation of long standing alkaptonuria, through careful radiological, physical, and laboratory examination. In this report, we describe 4 cases of ochronotic arthropathy to which we applied cementless total hip prosthesis due to severe hip involvement.

Stress hormones during the wake-up test in scoliosis surgery.

STUDY OBJECTIVE: To compare hemodynamic and endocrine stress responses of two anesthetic regimes during intraoperative wake-up tests in scoliosis surgery. DESIGN: Randomized, controlled clinical study. SETTING: University hospital. PATIENTS: 40 ASA physical status I and II teenage patients scheduled for scoliosis surgery. INTERVENTION: Patients were randomly divided into two groups: the propofol group (Group P) and the sevoflurane group (Group S). In Group P, anesthesia was induced with propofol and remifentanil and was maintained with infusions. Sevoflurane and remifentanil were used in Group S. After surgical instrumentation, patients were awakened, and the wake-up times were recorded. To determine the stress responses, blood samples were taken before induction, 10 minutes after surgical incision, before the wake-up test, during the wake-up test, and 10 minutes after the wake-up test. Cortisol, epinephrine, and norepinephrine concentrations, and hemodynamics all were recorded at the same time. MEASUREMENTS AND MAIN RESULTS: The times from discontinuation of anesthetics to eye opening and movement were similar in both groups. Epinephrine and norepinephrine concentrations during the wake-up test were significantly higher than pretest results in both groups (p < 0.001). There were no statistically significant differences between groups in heart rate or blood pressure. CONCLUSIONS: Propofol-remifentanil anesthesia is equivalent to sevoflurane-remifentanil anesthesia for a wake-up test. Both the propofol- and sevoflurane-based anesthetic regimens abolish hemodynamic and endocrine stress responses to incision for scoliosis surgery in teenagers. Intraoperative wake-up testing is associated with substantial catecholamine stress despite virtually unchanged mean arterial pressure and heart rate.

Anterior tibial compartment syndrome due to the pyomyositis in a patient with rheumatoid arthritis. A case report.

Anterior tibial compartment syndrome was developed due to pyomyositis in a 33-year-old male patient with rheumatoid arthritis while receiving steroid therapy during the follow-up period. The preoperative physical examination, laboratory findings, MRI images, intraoperative observation and postoperative histopathological examinations confirmed the association with pyomyositis. The surgical drainage and antibiotic treatment were effective, and in the follow-up period, neuromuscular dysfunctions disappeared completely within 6 months. The patient has been asymptomatic for 4 years of follow-up. To date, anterior tibial compartment syndrome due to pyomyositis in a case with rheumatoid arthritis has not been reported.

A case of I-cell disease (mucolipidosis II) presenting with craniosynostosis.

CASE REPORT: In this paper, a patient with mucolipidosis II (I-cell disease) is described. The initial findings were microcephaly and metopic craniosynostosis. He had coarse facial features and dysostosis multiplex. The first child in his family had died with severe bone pathology at 5 months of age. CONCLUSION: The case is presented to emphasize that craniosynostosis may be the first symptom in mucolipidosis II.